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| CASE REPORT |
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| Year : 2019 | Volume
: 6
| Issue : 1 | Page : 33-36 |
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Exogenous ochronosis in an elderly Indian male: A case report
Chapalamadugu S Sindhu, Cordelia Babitha, Anuradha Priyadarshini, Mahalakshmi Veeraraghavan
Department of Dermatology, Sri Ramachandra University, Chennai, Tamil Nadu, India
| Date of Web Publication | 4-Jul-2019 |
Correspondence Address:
Chapalamadugu S Sindhu
Department of Dermatology, Sri Ramachandra University, Chennai, Tamil Nadu
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/Pigmentinternational.Pigmentinternational_
Exogenous ochronosis (EO) is an infrequent cutaneous disorder characterized by blue-black hue in the skin due to the deposition of small ochre-colored pigment in the dermis secondary to prolonged skin lightening agents or unprotected sun exposure. We describe a case of a 65-year-old male who presented to our hospital with a history of hydroquinone application for melasma and was diagnosed to have acquired ochronosis after subsequent extensive history taking, clinical, dermoscopic, and histopathological assessment. EO is not frequently reported as it is misdiagnosed as melasma in most of the Asian population. In such a situation, histopathological examination of the skin lesion remains a gold standard for diagnosis of the condition. EO is a cosmetic disfigurement over the face with unsatisfying treatment options.
Keywords: Exogenous ochronosis, hydroquinone, hyperpigmentation, maculopapules, melasma
How to cite this article:
Sindhu CS, Babitha C, Priyadarshini A, Veeraraghavan M. Exogenous ochronosis in an elderly Indian male: A case report. Pigment Int 2019;6:33-6 |
| Introduction | |  |
Exogenous ochronosis (EO) is an infrequent symmetrically progressive cutaneous disorder distinguished by blue-black hue in the skin due to the deposition of small ochre-colored pigment in the dermis secondary to either the topical application of hydroquinone (HQ: 1,4-benzenediol), phenol, resorcinol, mercurials, and picric acid or to the administration of quinine, antimalarials or by sun exposure.[1] EO was first described by Beddard and Plumtrein following the use of phenol for leg ulcers. The prolonged exposure of HQ (usually beyond 3%) was first demonstrated by Findlay (1975).[8] It manifests as paradoxical hyperpigmentation displaying gray-brown or blue-black macules (hyperchromic, pinpoint, caviar-like papules) and transpires at the osseous surfaces of the zygomatic areas of the face, side, and back of the neck.[3] It predominantly occurs at the region of chemical contact in a symmetrical pattern and is a cosmetically disfiguring and psychologically disturbing condition. The incidence of EO globally remains unknown but is relatively low in Asia with increasing reports especially from India, China, Thailand, and Singapore.[4] A South African epidemiological study by Hardwick et al.[5] showed that the prevalence of EO is 70% with skin lightener users and most importantly the gender preponderance is with female (42%) than males (15%) between the age group of 40 to 49 years. At present, most of the case report literature from India are of female gender and only a single report of a 45-year-old male has been documented.[6] Hence, we report a case of an elderly aged male who had presented to our hospital with a history of HQ application for melasma and was diagnosed to have acquired ochronosis or EO.
| Case report | | |
A 65-year-old male presented to our hospital with a complaint of progressive pigmentation of the face for 10 years. He had been applying the combination of topical HQ (3%), octinoxate (octyl methoxycinnamate) and oxybenzone (benzonephenone-3) on a daily basis, which had been advised for a month but he happened to continue the same for 10 years. There was no history of pigmentation elsewhere in the body. There was no history of dark colored urine at anytime. He had no history of itching, photosensitivity, drug intake, smoking, alcoholism, or emotional stress. He had no history of joint and chest pain. He is a known diabetic and hypertensive and is on regular medication for the same.
Physical examination revealed blue-black maculopapular lesions symmetrically distributed over the face involving the zygomatic region and the nose [[Figure 1] and [Figure 2]]. | Figure 1 Demonstration of blue-black maculopapular lesions over the zygomatic region
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Routine investigations like complete blood count, urinalysis, renal function test, liver function test, serum uric acid, tests for thyroid function, urine for homogentisic acid, antinuclear antibodies, and electrocardiography did not show any abnormality. Increased blood sugar levels confirmed his state of diabetes. Dermoscopy revealed amorphous blue-gray areas on a background of brown pigmentation with perifollicular halo and white dots [[Figure 3]]. Histopathological examination of the skin lesion revealed golden brown colored structures intermingled between dermal collagen (particularly from upper to mid dermis) along with a few pigmented granular forms. The analysis also showed the presence of small lymphocyte aggregates at the papillary dermis and a relatively normal epidermis. Hence, the histopathological features were consistent with the diagnosis of EO [[Figure 4] and [Figure 5]]. | Figure 3 Dermoscopy of the ochronotic lesion showing amorphous blue-gray areas with perifollicular halo
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| Discussion | | |
The etiological factors behind EO have been extensively studied by Hull and Procter[7] and include use of prolonged skin lightening agents (predominantly HQ) and unprotected sun exposure. The exact molecular mechanism of the predisposing factor of EO is unknown. Literature published by Findlay et al. explained the key mechanism in the causation of EO as inhibition of melanin biosynthesis by HQ (industrial hyrdroxyphenolic compound) through the blockage of the enzyme tyrosinase, thus hampering DNA and RNA synthesis.[8],[9] Penneys[10] proposed that the hyperpigmentation was a result of homogentisic acid oxidase enzyme inhibition by HQ leading to the local accumulation of homogentisic acid in the skin which undergoes polymerization to form ochronotic pigment. EO manifests as asymptomatic gray-brown or blue-black macules over the malar areas, temples, and neck.[3] Histopathological examination reveals yellow-brown or ochre colored banana-shaped fibers in the papillary dermis. Swelling and homogenization of the collagen bundles may be noted in the papillary and reticular dermis.[4] The clinical and histopathological findings of our report had demonstrated that the EO was a result of prolonged application of topical HQ and the mechanism might involve any one of the pathways mentioned above.[8],[10]
The potential effect of HQ is directly related to its applied concentration at the site. Generally, a 2% HQ cream or an alcohol-based preparation is employed for clinical treatment and has been observed to be safe. Further the frequency and duration of application of topical 2% HQ also affects the absorption of the drug into the skin and may cause EO.[2],[11]
The six major Indian case reports have highlighted the importance of the skin lightening (HQ 2%–5%) application as a major cause of EO and that too predominantly in females.[12],[13],[14] A lone descriptive study by Kulandaisamy et al.[15] spanning 1.5 years has evaluated 104 melasma patients in which 10 male (40.4 ± 9.3 years) patients were documented to have EO secondary to HQ application for melasma. This could probably be due to the fact that females tend to be more conscious about their appearance and thus seek medical treatment. Further hormonal factors play a role in melasma which is more pronounced in females and less likely in males and hence males might be at a lesser chance of developing EO. There are only a few reports of EO in males and we would like to highlight that our case is an elderly male reported to develop EO.
Treatment modalities for EO include strict sun protection with physical sunblocks or chemical sunscreens, topical retinoic acid and glycolic acid and chemical peeling with glycolic acid. Q-switched alexandrite and Q-switched ruby laser also enhanced the clearance of EO.[4]
| Conclusion | | |
Patients often reach a dermatologist for pigmentary conditions, especially melasma. With an overenthusiastic approach to apply the prescribed cream(s), especially knowing that they would act to lighten the skin, patients do not realize the possibility of developing side effects such as EO. EO on the other hand is a frequently missed entity resulting as a consequence of relentless use of HQ with disappointing treatment options. Hence, it is often prudent to educate patients regarding the conscious and controlled usage of HQ-based preparations.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his names and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
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