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Gastroentérologie Clinique et Biologique
Vol 31, N° 2  - février 2007
pp. 166-168
Doi : GCB-02-2007-31-2-0399-8320-101019-200520008
An uncommon perianal nodule: Hidradenoma papilliferum
 

Fady Daniel [1], Amel Mahmoudi [2], Vincent de Parades [2], Jean-Francois FlÉjou [3], Patrick Atienza [2]
[1] Unité de Proctologie, Service de Gastroentérologie, CHU Hôtel-Dieu de France, Université Saint Joseph, boulevard Alfred Naccache, Beyrouth, Liban
[2] Service de Proctologie, Groupe hospitalier Diaconesses — Croix Saint-Simon, Paris
[3] Service d'Anatomie-Pathologique, Hôpital Saint-Antoine, AP-HP, Paris.

Tirés à part : V. de Parades [1]

[1] Service de Proctologie, Groupe hospitalier Diaconesses — Croix Saint-Simon, 18, rue du Sergent Bauchat, 75012 Paris.

proctologie@hopital-dcss.org

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L'hidradénome papillifère: une tumeur rare de la région périanale

L'hidradénome papillifère est une tumeur bénigne kystique des glandes apocrines, survenant presque exclusivement chez les femmes au niveau de l'épiderme de la peau de la région anogénitale. Nous présentons le cas d'une malade de 66 ans qui s'est plainte d'une tuméfaction anale augmentant progressivement de volume. Par la suite, nous discutons les aspects clinico-histologiques de cette entité ainsi que le diagnostic différentiel à la lumière des données de la littérature. Une excision chirurgicale a été nécessaire pour l'identification. Aucune récidive n'a été observée après un an de suivi.

Abstract

Hidradenoma papilliferum is a rare, benign, cystic, papillary apocrine gland tumor that occurs almost exclusively in women in the skin of the anogenital region. We present the case of a 66 year old woman who presented with a progressively enlarging perianal nodule. We provide a review of the literature emphasizing the clinico-pathological characteristics and differential diagnosis. Local excision is necessary for identification and cure. No recurrence was observed after one year follow-up.


Introduction

Hidradenoma papilliferum (HP) is a benign, cystic, papillary apocrine gland tumor that occurs almost exclusively in women in anogenital region [1]. The clinical characteristics of this tumor may be confusing, since it is uncommon. We present the case of a 66 year old woman who presented with a progressively enlarging perianal nodule. We provide a review of the literature describing the clinico-pathological characteristics and differential diagnosis. Local excision is necessary for identification and cure. No recurrence was observed after one year follow-up.

Case report

A 66 year old woman consulted for a painless enlarging perianal nodule. Her past history was unremarkable except for hysterectomy for uterine fibromatosis. She had first noticed the nodule one year before. She was concerned because it had progressively enlarged over the last few weeks. There was no history of pain, itching, bleeding, fever or purulent discharge. Proctological examination revealed a 1.5 cm firm, freely mobile non-tender bluish nodule, located in the right anterior quadrant of the anal margin (figure 1). Gynecological examination and anuscopy were normal. Anal endosonography showed a 15x9 mm hypoechoeic subcutaneous, cystic nodule with ill defined margins. The content was heterogeneous with posterior reinforcement. The sphincteric apparatus was normal, with no evidence of collection or fistula (figure 2). The nodule was excised under local anesthesia (figure 3). Histological analysis disclosed a benign cystic lesion with papillary folds lined with columnar eosinophilic cells presenting apocrine differentiation, providing a diagnosis of Hidradenoma papilliferum (HP) (figure 4 and 5). The patient showed no evidence of recurrence after one year follow-up.

Discussion

HP is an uncommon benign tumor, originating in the apocrine sweat glands [1]. It is located almost exclusively in the vulvar and anal areas. It occurs four times more frequently on the vulva than in the perianal area. Patients are usually women from 30 to 60 years old [2]. Only one case has been described in a male patient [3]. The average age of patients with perianal lesions is higher than that of patients with vulvar involvement [4]. Extragenital localization qualified as ectopic HP is quite rare and has been described on the scalp [5], eyelids [6 et 7], face [8], external auditory canal [9], auricular area [10], nasal skin [11], the trunk [12 et 13], the limbs and axilla [14]. Ectopic HP are slightly more frequent in women ranging from (8-78 years old) [14].

The lesion is usually asymptomatic, but may be revealed by itching, pain, bleeding or discharge, especially if it ulcerates. It is typically smaller than 2 cm, but reports of tumors up to 4 cm have been published [2]. It presents as a solitary, firm, freely mobile well demarcated nodule covered by normal skin, as in our case. Similar to its anogenital counterpart, most ectopic lesions are asymptomatic and grow for a long time before resection. Occasional reports on malignization of anogenital HP exist and it has been misdiagnosed as an adernocarcinoma [15 et 16]. In this context, mitotic figures, cellular pleomorphism or atypia, suggest malignant transformation. Nevertheless, all ectopic lesions behave in a benign fashion and do not recur or metastasize [13].

Histopathologic examination shows a well-circumscribed, cystic lesion in the mid dermis surrounded by a fibrous capsule with no connection to the overlying epidermis. Within the tumor, prominent papillary folds project into the cystic cavity. The lumen is lined with a single layer or multiple rows of columnar cells, with an oval, pale-staining nucleus near the base and evidence of active “decapitation” secretion like that seen in apocrine glands [1]. The absence of connective tissue in the papillary structures distinguishes HP from syringocystadenoma papilliferum [1]. The latter usually occurs in the head and neck region, either de novo or within a nevus sebaceus. It shows duct-like invaginations and cyst-like cavities of various shapes and sizes extending from the epidermal surface into the body of the lesion. These extensions characteristically have keratinized epithelium near the surface of the lesion transitioning to ducts lined by two epithelial cell layers, with an inner columnar layer resting on a basal low-cuboidal epithelial layer [17]. The connective tissue stroma surrounding these duct-like structures contains a chronic inflammatory infiltrate with plasma-lymphocytic predominance [18]. In contrast, HP originates below the level of the epidermis, lacks epidermal surface invaginations and plasma cell infiltrates, and more often has a cystic architecture. Lesions with mixed features have been described [19].

The spectrum of differential diagnosis in case of HP is wide, comprising most commonly thrombosed external hemorrhoids, anal abscess, viral warts, and epidermoid carcinoma. Less often, it may be confused with a sebaceous cyst, lipoma, or neurofibroma, or in case of associated pigmentation, anal endometriosis, angioma or melanoma [1 et 2]. Treatment relies on surgical resection which establishes the diagnosis after pathologic examination. Recurrence is rare [1, 2, 3, 4, 5, 6 et 7] [8, 9, 10, 11, 12, 13 et 14].

In conclusion, HP is an uncommon benign anal tumor usually occurring in women Awareness of this entity is important for the proctologist, because it lacks distinctive clinical characteristics. Diagnosis is based on histopathology. Surgical excision is therefore required for identification and definitive cure.

Références

[1]
Elder D, Elenitsas R, Ragsdale BD. Hidradenoma papilliferum. In: Elder D, Elenitsas R, Jaworsky C, Johnson B Jr, eds. Lever's histopathology of the skin. 8th ed. Philadelphia: Lippincott-Raven; 1997: 770-1.
[2]
Handa Y, Yamanaka N, Inagaki H, Tomita Y. Large ulcerated perianal Hidradenoma papilliferum in a young female. Dermatol Surg 2003; 29: 790-2.
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Marrogi AJ, Wick MR, Dehner LP. Benign cutaneous adnexal tumors in childhood and young adults, excluding pilomatrixoma: review of 28 cases and literature. J Cutan Pathol 1991; 18: 20-7.
Vanatta PR, Bangert JL, Freeman RG. Syringocystadenoma papilliferum. A plasmacytotropic tumor. Am J Surg Pathol 1985; 9: 678-83.
Schewach-Millet M, Trau H. Congenital papillated apocrine cystadenoma: a mixed form of hidrocystoma, Hidradenoma papilliferum, and syringocystadenoma papilliferum. J Am Acad Dermatol 1984; 11: 374-6.




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