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CASE REPORT
Year : 1996  |  Volume : 44  |  Issue : 3  |  Page : 168-169

An unusual case of syringocystadenoma papilliferum of the eyelid


Department of Ophthalmology, JIPMER, Pondicherry, India

Correspondence Address:
Vasudev Anand Rao
Department of Ophthalmology, JIPMER, Pondicherry-605 006
India
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Source of Support: None, Conflict of Interest: None


PMID: 9018997

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How to cite this article:
Rao VA, Kamath GG, Kumar A. An unusual case of syringocystadenoma papilliferum of the eyelid. Indian J Ophthalmol 1996;44:168-9

How to cite this URL:
Rao VA, Kamath GG, Kumar A. An unusual case of syringocystadenoma papilliferum of the eyelid. Indian J Ophthalmol [serial online] 1996 [cited 2019 Aug 26];44:168-9. Available from: http://www.ijo.in/text.asp?1996/44/3/168/24580

Syringocystadenoma papilliferum, formerly known as naevus syringocystadenomatous papilliferus is a benign adnexal tumour of the skin.[1] We report an unusual case of this entity occurring on the eyelid.


  Case report Top


A 45 year old woman presented with a lesion on the medial aspect of right upper eyelid. She had a small elevated nodular swelling at the margin of the eyelid for the past three decades. There was gradual increase in the size of the swelling for past 8 years and over past 6 months it became more noticeable combined with discomfort, itching and bleeding on manipulation.

Physical examination revealed a solitary 1.5 x 2 cm size brownish firm fleshy growth with hyperkeratotic surface covered with crusts in some areas, involving the skin of the right upper eyelid and over-hanging the inner canthus [Figure - 1]. The upper lid margin was involved but not beyond the lash line. It did not involve the underlying deeper tissues also and there was no regional lymphadenopathy. The remainder of ocular and general physical examination was unremarkable.

Though the clinical suspicion was either a squamous cell or a basal cell carcinoma, the pathologic diagnosis of a small wedge biopsy specimen was syringocystadenoma papilliferum with no evidence of malignancy. The mass was excised along with a 3mm margin of healthy skin and the resultant skin defect was repaired with a median frontoglabellar pedicle skin flap. The final pathologic diagnosis of excision biopsy specimen concurred with the initial report.


  Pathologic Findings Top


Histopathologic examination demonstrated cystic invaginations extending downward from the epidermis [Figure - 2]. Numerous papillary projections extended into the lumen of the invagination. The papillary projections were lined by two rows of cells, an inner row of columnar cells demonstrating active "decapitation" secretion and an outer row of small cuboid cells. A dense dermal cellular infiltrate consisting primarily of plasma cells was prominent in papillary projections.


  Discussion Top


Syringocystadenoma papilliferum is an uncommon skin adnexal tumour that occurs as a solitary lesion. Although most lesions are not clinically distinctive and require biopsy for diagnosis, the tumour is usually described as a skin coloured to pink, hairless, firm plaque of grouped nodules or as a solitary nodule.[2] Cauliflowerlike, verrucous, papillary, hyperkeratotic or sometimes moist fleshy excrescences have also been described.[3] Some tumours may show central umblication through which small fistulae may discharge fluid.

Most of the lesions develop and enlarge gradually, although a few can attain considerable size within a few months. Scratching to relieve itching causes irritation and bleeding. The ulcers that form in such irritated lesions tend to crust and breakdown repeatedly as occurred in our case.

In about 75% of cases, syringocystadenoma papilliferum arises within a preexistent organoid sebaceous naevus of Jadassohn.[3] Such a lesion, usually the plaque variety is present from birth in the scalp, face or on the neck, which at puberty becomes more elevated and nodular, verrucous or crested. The remaining 25% of syringocystadenoma papilliferum arise on the trunk and genitocrural region during adolescence or adult life without a preexisting lesion, usually in the solitary nodular form. Our present lid lesion arose in this manner.

The occurrence of syringocystadenoma papilliferum in the eyelids is very rare. In a large series of 100 cases reported by Helwig and Hackney,[3] 55 were on the scalp, 11 on the forehead and temple, 5 on the face and 3 on the upper lip, but none was reported on the eyelid. To our knowledge only two cases are reported by Jakobiec et al.[4]

Though transition to carcinoma is rare, it has occurred. In about one-tenth of cases of syringocystadenoma papilliferum, basal cell carcinoma can secondarily develop.[3] For this reason surgical excision is the treatment of choice for this tumour. Other associated complications of this tumour include ulceration and secondary infection. We performed excision of the tumour and the resultant skin defect was repaired with median frontoglablellar pedicle skin flap. The patient was asymptomatic at 18 months followup.

 
  References Top

1.
Pinkus H. Life history of neavus syringo adenomatosus papilliferus. Arch Dermatol 69:305-322, 1954.  Back to cited text no. 1
    
2.
Rook A, Wilkinson DS, Ebling FJG, et al. Textbook of Dermatology, Blackwell Pondicherry 1986:177.  Back to cited text no. 2
    
3.
Helwig EB, Hackney VC. Syringoadenoma papilliferus. Lesion with and without naevus sebaceus and basal cell carcinoma. Arch Dermatol 71:361-372, 1955.  Back to cited text no. 3
    
4.
Jakobiec FA, Streeten BW, Iwamoto T, et al. Syringocystadenoma papilliferum of the eyelid. Ophthalmology 88:1175-1181, 1981.  Back to cited text no. 4
    


    Figures

  [Figure - 1], [Figure - 2]



 

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