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E-IJD-CASE REPORT
Year : 2015  |  Volume : 60  |  Issue : 1  |  Page : 104
Eruptive collagenoma: A rarely reported entity in Indian literature


Department of Dermatology, Venereology and Leprology, Goa Medical College, Bambolim Goa, India

Date of Web Publication26-Dec-2014

Correspondence Address:
Joycelin Fernandes
Department of Dermatology, Venereology and Leprology, Goa Medical College, Bambolim Goa
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.147853

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   Abstract 

Eruptive collagenomas are non familial connective tissue nevi of unknown etiology presented with an abrupt onset. While most cases are reported in young adults, there is a paucity of literature in children. We report a case of a 4-year-old girl, who presented with multiple asymptomatic, papules, plaques and nodules on the face, trunk and upper extremities with no systemic involvement. Histopathologically, the lesion showed thickened homogenized collagen fibres highlighted by Masson's trichrome stain and paucity in elastic fibres by Verhoeff-van Gieson stain, confirming the diagnosis of eruptive collagenoma.


Keywords: Child, eruptive collagenoma, face


How to cite this article:
Barad P, Fernandes J, Shukla P. Eruptive collagenoma: A rarely reported entity in Indian literature. Indian J Dermatol 2015;60:104

How to cite this URL:
Barad P, Fernandes J, Shukla P. Eruptive collagenoma: A rarely reported entity in Indian literature. Indian J Dermatol [serial online] 2015 [cited 2020 Sep 26];60:104. Available from: http://www.e-ijd.org/text.asp?2015/60/1/104/147853

What was known?
Eruptive Collagenomas which are hamartomas of collagen, although known to occur in the first two decades of life, are usually reported in adult life, with lesions predominantly on the trunk and extremities.



   Introduction Top


Connective tissue nevi of the skin are hamartomas, consisting predominantly from one of the components of the extracellular matrix namely collagen, elastic fibres or proteoglycans. [1] Those predominantly composed of collagen are called collagenomas. We hereby describe a case of eruptive collagenoma in a child which started at an early age of three and half years. We report this case due to its relative paucity of literature in paediatric population coupled with appearance of lesions over an atypical site (face).


   Case Report Top


A 4 year old girl presented to the dermatology department with history of noticing multiple, grouped, asymptomatic raised skin lesions predominantly in bilateral axillae, upper back and face for 6 m. The lesions appeared on normal skin, without any preceding inflammation or trauma. There was no history of epilepsy, developmental anomalies, delay in milestones or any pertinent family history.

Clinical examination revealed multiple, symmetrically distributed, brownish papules and plaques having a leathery surface ranging in size from 0.2 × 0.2 cm [Figure 1] to 2.5 × 2.5 cm [Figure 2]. Few skin coloured, firm, non tender nodules, with surface showing irregular thickening, giving a peau d' orange appearance were noted in the axillae [Figure 3]. No neurocutaneous stigmata, thickened nerves or systemic abnormality was detected.
Figure 1: Well defined brownish plaques having a leathery surface on flexor aspect of left forearm

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Figure 2: Multiple brownish papules and plaques on the face

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Figure 3: Skin coloured nodules with surface showing irregular thickening giving a peau d' orange appearance

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Considering a differential diagnosis of connective tissue nevus, Histoid Hansen's and neurofibromatosis the patient was investigated.

Routine investigations were normal, while histopathology revealed a thickened reticular dermis with haphazardly arranged thickened collagen bundles suggestive of collagen naevus on haematoxylin and eosin stain [Figure 4]. Masson's trichrome highlighted an increase in the collagen tissue in the dermis [Figure 5], while Verhoeff-van Gieson showed a marked paucity of elastic fibres [Figure 6].
Figure 4: Skin biopsy revealed thickened reticular dermis with thick and haphazardly oriented collagen bundles. (H and E, x100)

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Figure 5: Photomicrograph showing dense thick collagen bundles in the dermis. (Masson's trichrome stain, x100)

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Figure 6: Photomicrograph demonstrating an increased collagen with marked reduction in elastic fibres. (Verhoeff-van Gieson stain, x100)

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   Discussion Top


In an attempt to classify collagenomas, Uitto et al. characterized them, based on pattern of distribution (localized or generalized) and mode of inheritance (acquired or inherited). While inherited autosomal dominant collagenomas include familial cutaneous collagenomas and shagreen's patches of Tuberous Sclerosis, those that are acquired are eruptive collagenomas and isolated collagenomas. [2]

Eruptive collagenoma, first reported by Cramer in 1966, [3] presents with sudden symmetrical appearance of several firm skin-coloured papules and nodules of various sizes, usually less than 1 cm in diameter, [4] on the trunk and upper extremities, [5] in the first two decades of life. The incidence and pathogenesis is unknown with no established family history or associated systemic findings.

Histopathologically, the lesions are characterized by an excessive accumulation of randomly arranged dense collagen bundles with either diminished, altered or absent elastic tissue. [4]

Eruptive collagenoma should be differentiated from other collagenomas namely familial cutaneous collagenoma, which is an autosomal dominated disorder, first described by Henderson et al., in 1968, characterized by clinical features similar to eruptive collagenoma. However, there is a positive family history, third decade presentation and associated systemic involvement. [6]

Isolated Collagenomas are sporadically acquired collagenomas localized to one body region and not associated with any disease. [7] They can present as paving stone naevi, plantar collagenoma, zosteriform lesions and papulolinear lesions. [8]

Shagreen patch is a collagenoma variant associated with Tuberous sclerosis, characterized by single to few asymmetrically distributed skin-coloured plaques of variable size predominantly in the lumbosacral region. [9]

Papular elastorrhexis and nevus anelasticus are closely related entities which mimic eruptive collagenomas both clinically and histologically. Papular elastorrhexis is characterised by multiple, 2 mm to 5 mm, flat, firm papules over the trunk and extremities while nevus anelasticus present as perifollicular papules. Both these conditions are associated with a decrease in elastic fibres. [10] Due to paucity of reported literature it is uncertain, whether these entities reflect a part of the same disease process or are separate disease states.

This patient was diagnosed as eruptive collagenoma based on first decade presentation of multiple lesions, histopathological findings and the absence of family history and systemic findings.

This child first presented with lesions at 3 1 / 2 years of age. Though in most cases the onset of presentation has been beyond the first decade, [5],[11],[12],[13],[14] only two cases have been reported with development of lesions within the first decade. [15],[16] The earliest onset of presentation was reported by Yahya et al., [15] in a 2 year old Nigerian girl and the other by Lee et al., [16] with onset at 5 years. Only 2 case reports by Mukhi et al. [12] and Yahya et al. [15] report involvement of the face in addition to other more common sites of presentation.

An extensive review of Indian literature has yielded a paucity of case reports of eruptive collagenoma. Although this entity is relatively uncommon, a good clinical acumen and a degree of suspicion help in the clinical diagnosis of such cases, while histopathology confirms the diagnosis.

 
   References Top

1.
Uitto J, Santa Cruz DJ, Eisen AZ. Connective tissue nevi of the skin. Clinical, genetic and histopathologic classification of hamartomas of the collagen, elastin and proteoglycan type. J Am Acad Dermatol 1980;3:441-61.  Back to cited text no. 1
    
2.
Uitto J, Santa-Cruz DJ, Eisen AZ. Familial cutaneous collagenoma: Genetic studies on a family. Br J Dermatol 1979;101:185-95.  Back to cited text no. 2
    
3.
Cramer HJ. On the clinical aspects of the so-called "eruptive collagenoma". Hautarzt 1966;17:437-40.  Back to cited text no. 3
    
4.
de Almeida HL Jr, Breunig Jde A, Wolter M, de Castro LA, Rocha NM. Light and electron microscopy of eruptive collagenoma. J Cutan Pathol 2009;36:35-8.  Back to cited text no. 4
    
5.
Xia Y, Darling TN. Rapidly growing collagenomas in multiple endocrine neoplasia type I. J Am Acad Dermatol 2007;56:877-80.  Back to cited text no. 5
    
6.
Henderson RR, Wheeler CE Jr, Abele DC. Familial cutaneous collagenoma. Arch Dermatol 1968;98:23-7.  Back to cited text no. 6
    
7.
Ishii N, Maeyama Y, Nakama T, Hashimoto T. A case of solitary collagenoma localized on the upper lip mimicking mucocele. Clin Exp Dermatol 2009;34:243-4.  Back to cited text no. 7
    
8.
Lo LK, Tsai TF, Chen YF, Hung CM, Ko WC. Papulo-linear collagenoma with arborizing arrangement: Report of a case. Pediatr Dermatol 2009;26:111-2.  Back to cited text no. 8
    
9.
Morgan JE, Wolfort F. The early history of tuberous sclerosis. Arch Dermatol 1979;115:1317-9.  Back to cited text no. 9
    
10.
Ryder HF, Antaya RJ. Nevus anelasticus, papular elastorrhexis, and eruptive collagenoma: Clinically similar entities with focal absence of elastic fibers in childhood. Pediatr Dermatol 2005;22:153-7.  Back to cited text no. 10
    
11.
McClung AA, Blumberg MA, Huttenbach Y, Colome-Grimmer MI, Raimer SS. Development of collagenomas during pregnancy. J Am Acad Dermatol 2005;53:S150-3.  Back to cited text no. 11
    
12.
Mukhi SV, Kumar P, Yuvarajkumar D, Raghuveer CV. Eruptive collagenoma. Indian J Dermatol Venereol Leprol 2002;68:98-9.  Back to cited text no. 12
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13.
Smith LR, Bernstein BD. Eruptive collagenoma. Arch Dermatol 1978;114:1710-1.  Back to cited text no. 13
    
14.
Batra P, Loyd A, Patel R, Walters R, Stein JA. Eruptive collagenomas. Dermatol Online J 2010;16:3.  Back to cited text no. 14
    
15.
Yahya H, Rafindadi AH. Eruptive collagenoma in a Nigerian girl. Int J Dermatol 2006;45:1344-6.  Back to cited text no. 15
    
16.
Lee MW, Choi JH, Sung KJ, Moon KC, Koh JK. A case of eruptive collagenoma. Pediatr Dermatol 2002;19:565-7.  Back to cited text no. 16
    

What is new?
There is a relative paucity of published case reports in pediatric age group. To the best of our knowledge and after extensive review of literature, we could not find any case reports in Indian pediatric population. Besides this, involvement of atypical sites like the face is also not commonly reported.


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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