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Year : 2020  |  Volume : 21  |  Issue : 2  |  Page : 126-131

Orofacial pigmentary lesions in the pediatric age group: A descriptive observational study

1 Department of Dermatology, Base Hospital, Delhi Cantt., New Delhi, India
2 Department of Dermatology, MLN Medical College, Allahabad, Uttar Pradesh, India
3 Department of Dermatology, AFMC, Pune, Maharashtra, India
4 Department of Dermatology, Base Hospital, Guwahati, Assam, India

Date of Submission16-Feb-2019
Date of Decision13-Mar-2019
Date of Acceptance01-Jan-2012
Date of Web Publication01-Apr-2020

Correspondence Address:
Sushil Kumar
Department of Dermatology, MLN Medical College, Allahabad, Uttar Pradesh
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijpd.IJPD_18_19

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Background: Pigmentary disorders on the face are frequently encountered in all age groups and both genders but are more common in pediatric patients. These lesions can be isolated incidental findings or indicative of a congenital or acquired systemic or genetic disorder. Aims and Objectives: The aim and objective of this study is to study the frequency and pattern of pigmented orofacial lesions in the pediatric age group and to describe the underlying associated condition if any. Materials and Methods: A descriptive study was conducted over a period of 6 months (June–November 2018) in the dermatology department of a tertiary care hospital in Eastern Uttar Pradesh. All the children from 2 to 14 years of age who reported to us with complaints of pigmentary lesions on the face or oral cavity were included, and attempt was made to find out if any underlying conditions. Results: A total of 207 children were included in the study. Pityriasis alba was the most common pigmentary disorder on the face in the study population in 146 (70.5%) children, while pityriasis versicolor was noted in 21 (10.1%). Other facial dermatoses noted were vitiligo, congenital melanocytic nevus, and postinflammatory hypopigmentation and hyperpigmentation. Eight (3.8%) children had associated underlying diseases such as xeroderma pigmentosum, dyskeratosis congenita, Vitamin B12 deficiency, Laugier–Hunziker syndrome, and juvenile dermatomyositis. Conclusions: Pigmentary disorders of the face and oral cavity are frequently seen in children which forms a major part of dermatology consultation. Although most of these disorders are of cosmetic concerns, few conditions are associated with systemic conditions. A thorough investigation and management are the keys to manage such cases.

Keywords: Pigmentary disorders, pityriasis alba, xeroderma pigmentosa

How to cite this article:
Saraswat N, Chopra A, Kumar S, Neema S, Kamboj P. Orofacial pigmentary lesions in the pediatric age group: A descriptive observational study. Indian J Paediatr Dermatol 2020;21:126-31

How to cite this URL:
Saraswat N, Chopra A, Kumar S, Neema S, Kamboj P. Orofacial pigmentary lesions in the pediatric age group: A descriptive observational study. Indian J Paediatr Dermatol [serial online] 2020 [cited 2020 Dec 4];21:126-31. Available from: https://www.ijpd.in/text.asp?2020/21/2/126/281732

  Introduction Top

Pigmentary disorders are commonly encountered in all age groups and are a frequent cause of consultation in the pediatric age group and adolescents.[1] The presence of pigmentary lesions on the face or oral cavity is a cause of concern and anxiety among parents which brings these patients for dermatology consultation more often than adults.[2] Both hypopigmentary and hyperpigmentary lesions are seen in children and are mostly of cosmetic concern, but at times, these pigmentary lesions can have an underlying systemic or congenital disease associated. Although pigmentary dermatosis has been studied in the past in pediatric patients, studies describing pigmentary lesions on the face and oral mucosae are lacking. This study was conducted to bridge the existing gap in our understanding of frequency and association of facial pigmentary lesions in the pediatric age group.

  Materials and Methods Top

This study was conducted at a tertiary care hospital in Eastern Uttar Pradesh (UP) over a period of 6 months (June 2018–November 2018). Permission of the Institutional Ethics Committee was obtained. Convenience sampling was adopted, thus including all children between 2 and 14 years of age affected with pigmentary lesions on the face or oral cavity visiting the skin outpatient department (OPD) during our study were included in the study. Written informed consent from the parents after explaining the nature of the study was obtained. Patients, where permission could not be obtained, were excluded from the study. A detailed history was obtained from parents/patients regarding onset, progression, associated complaints, developmental milestones, drug intake, muscle weaknesses, joint swelling or tenderness, family history, and any other systemic complaints. A thorough dermatological examination was carried out in the natural daylight. Systemic examination was carried out in all the cases after obtaining a relevant history. Family members were also examined in case they had same complaints. Relevant investigations, such as examination of sensations on the hypopigmented patch, 10% potassium hydroxide mount for fungal infection, slit-skin smear for acid-fast bacilli in leprosy, woods lamp examination, diascopy, complete blood count, to rule out nutritional deficiencies and hematological abnormalities, were done wherever warranted. Sonography of the abdomen, barium meal, endoscopy, X-ray joints, creatine kinase level and magnetic resonance imaging (MRI) of relevant muscles were done when systemic disease or any syndrome was suspected. Data included age, gender, onset of complaints, associated complaints if any, any illnesses and family history were endorsed in predesigned pro forma. Data were analyzed using the SPSS software version 20.0 (IBM SPSS statistics for windows, IBM Corp., Armonk, NY, USA). Institutional Ethical Committee's permission was obtained.

  Results Top

A total of 207 children between the age of 2 and 14 years were studied for orofacial pigmentary lesions during the study period of 6 months at a tertiary care hospital in Eastern Uttar Pradesh. Among them, 109 (52.6%) were male, whereas 98 (47.3%) were female. The male-to-female ratio was 1.1. Pityriasis alba was the most common facial pigmentary condition we noted in 146 (70.5%) children followed by pityriasis versicolor in 21 (10.1%). Other facial pigmentary conditions noted included postinflammatory hypopigmentation in 9 (4.3%) children and postinflammatory hyperpigmentation in 7 (3.3%) children [Figure 1] and [Figure 2]. Vitiligo lesion on the face was noted in 6 (2.8%) children [Figure 3] and speckled nevus and pigmented verruca in 3 (1.4%) patients each [Figure 4] and [Figure 5]. Other dermatoses noted are depicted in [Table 1] and [Figure 6],[Figure 7],[Figure 8],[Figure 9],[Figure 10],[Figure 11],[Figure 12],[Figure 13],[Figure 14]. Eight (3.8%) cases of total 207 children studied had underlying associated systemic disease or genodermatosis, as shown in [Table 2]. Two patients in our study presented with multiple, well-defined brownish macules on the buccal and labial mucosa and gave a history of abdominal pain, bleeding per rectum, and altered bowel habits. These patients were found to have jejunal polyps on further investigations and were diagnosed to have Peutz–Jeghers syndrome. Another two children who had xeroderma pigmentosa presented with freckling on the face, whereas one had freckles on the forearms and neck also. There was a history of redness of the face on sun exposure in both while others gave a history of severe itching on the exposed part of the skin in the sun. One of these patients had corneal opacity. We also came across a female patient who had facial erythema along with a recurrent episode of periorbital edema. She gave a history of progressively increasing weakness of the upper and lower limbs. She had multiple episodes of joint pain and swelling along with fever for which she had consultation in her locality. Her antinuclear antibody (ANA) was positive, and creatine phosphokinase levels were increased. MRI of the thigh was suggestive of muscle involvement. She was diagnosed with a case of juvenile dermatomyositis (JDM). One patient who had mucosal hyperpigmentation along with freckles also had longitudinal melanonychia. All her relevant investigations were normal. The patient was diagnosed with a case of Laugier–Hunziker syndrome (LHS).
Figure 1: Postinflammatory hyperpigmentation in a child

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Figure 2: Postinflammatory hypopigmentation on the lips due to herpes labialis in a child

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Figure 3: Focal vitiligo on the right cheek of a child

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Figure 4: Speckled nevus in a child

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Figure 5: Speckled nevus

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Table 1: Spectrum of a facial pigmentary lesion noted in the study group

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Table 2: Underlying disorders/genodermatoses in the study participants

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Figure 6: Black-brown macules on tongue in a patient

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Figure 7: Congenital melanocytic nevi in a child

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Figure 8: Halo nevus in a child

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Figure 9: Mucosal pigmentation in Laugier–Hunziker syndrome

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Figure 10: Pigmented verruca

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Figure 11: Hyperpigmentation of the tongue in Vitamin B12 deficiency

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Figure 12: Facial erythema in a girl with juvenile dermatomyositis

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Figure 13: Freckles in a child

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Figure 14: Freckles in a girl child

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  Discussion Top

The epidemiology of pigmentary disorders is highly varied across the world, races and age groups. It is estimated that 6.9% of children and adolescence from North India suffer from pigmentary disorders, whereas in South India, the frequency of hypopigmentary and hyperpigmentary disorders is 3.28 and 1.54/1000, respectively, in children attending the dermatology OPD.[2],[3],[4] Pigmentary disorders were reported in 10.8% of the adult population attending dermatology OPD in Western India in a study.[5]

Pigmentary disorders, especially on the face, can have visual and psychological issues, more commonly in pediatric patients.[6] These disorders can be congenital or acquired and occasionally may be a manifestation of a serious underlying disorder or genodermatoses. The common pigmentary disorders encountered in pediatric patients include pityriasis alba, pityriasis versicolor, melanocytic nevus, vitiligo, postinflammatory hypopigmentation and hyperpigmentation, nevus depigmentosus, and freckles.[7],[8],[9] Epidemiology and frequency of facial pigmentary lesions and its underlying association have not been studied earlier which makes the present study unique. Oral pigmentary lesions can also be associated skin involvement or it can be a part of an underlying disorder and need to be carefully examined and investigated if required.

In the present study, the most common pigmentary lesions were pityriasis alba followed by pityriasis versicolor which is in alignment with earlier studies.[10],[11] In another study conducted by Pinto and Bolognia, the most common hypopigmented lesions in children were pityriasis alba, vitiligo, nevus depigmentosus, and pityriasis versicolor like in the present study.[12]

A history of atopy was obtained in 31 (21.2%) children out of 146 who had pityriasis alba which is similar to earlier studies where a personal history of atopy was given by 17% of patients. Low hemoglobin amounting to anemia was observed in only 19 (13%) children with pityriasis alba, whereas other studies have found that 15.5% of children with pityriasis had anemia.[13] Pityriasis alba mostly involves back and chest followed by the face in 15.87% children as seen in other studies; however, in the present study, we included the children with face involvement with or without the involvement of other parts of the body.[14]

The involvement of the face is quite common in pityriasis versicolor, and some studies have found that the face is most common area involved, however the present study was done only in children with involvement of the face.[15] Only 6 (2.8%) children gave a history of itching on these lesions, while in others, there were no associated complaints.

Postinflammatory hypopigmentation and hyperpigmentation was seen in 9 (4.3%) and 7 (3.3%) children, respectively, which is on the lower side as compared to earlier studies where these lesions were seen in 14% and 13% children, respectively. The cause for this variation could be explained by the fact that these studies were done to assess the cause of pigmentary lesions involving the whole body as compared to the present study where only facial skin was considered.[10]

Vitiligo patch was seen in 6 (2.8%) children, of which focal vitiligo was seen in 3, vitiligo vulgaris in 2 (0.9%), and acrofacial in one patient. The face has been described as the most common site of vitiligo by Jain et al. However, the most common pattern described is vitiligo vulgaris followed by focal vitiligo, segmental, mucosal, mixed, and acrofacial variety.[16] In the present study, we found that anemia was present in only one patient of vitiligo vulgaris, and there was no other autoimmune disease noted.

Speckled nevus was seen in 3 (1.4%) children in the present study, whereas pigmented verruca was seen in another 3 (1.4%) cases. Pigmented warts were exclusively involving the face in all three children, and there was no other body site involved. Congenital melanocytic nevus was seen in 2 (0.9%) children involving the face.

Of 207 children studied during the study period, 8 (3.8%) had associated genodermatosis or systemic disorder. Mucosal lentigines were seen in two children who were diagnosed to have Peutz–Jeghers syndrome. Three (1.4%) children had mucosal hyperpigmentation, of which one has associated acral pigmentation and was found to have Vitamin B12 deficiency on workup, other had dyskeratosis congenita. Another child who had hyperpigmented macules in the oral cavity and labial mucosa was suffering from LHS. Ephelids were seen in 2 (0.9%) children who were diagnosed to have xeroderma pigmentosus. A (0.4%) 12-year-old female presented with erythema on the face for the past 6 months along with recurrent episodes of fever and joint pain. She also had a history of muscle weakness, and ANA was positive. She was diagnosed with a case of JDM. Halo nevus was seen in 1 (0.4%) child on the face.

  Conclusions Top

Pigmentary lesions of the face or oral cavity are common in children and are associated with patient or parental anxiety. Although most of these lesions are just isolated clinical finding and do not require further workup except for counseling the parents or patients, few of these pigmentary anomalies are the manifestation of underlying systemic or congenital diseases and need to be investigated in detail. It is prudent for the dermatologists to be well versed with these disorders and to understand that at times only oral or facial involvement needs to be investigated to reach a diagnosis and treat that patient at the earliest.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patients had given their consent for their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

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  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14]

  [Table 1], [Table 2]


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