Sacral Nevus Lipomatosus Cutaneous Superficialis

Sir,

Nevus lipomatosus cutaneous superficialis (NLCS) is a rare type of connective tissue nevus characterized by the presence of mature adipose tissue in the dermis.[12] Clinically, NLCS is classified into two forms: multiple type and solitary type. The classical form is characterized by groups of multiple, soft, non-tender, pedunculated, cerebriform, yellowish or skin-coloured papules, nodules, or plaques.[3] A 32-year-old man admitted to hospital with multiple swelling over the sacrum [Figure 1]. The lesion had started as a small cutaneous mass when the patient was 3 years old. Lesions had increased slowly in numbers and size. He had no systemic complaints. There was no history of similar lesions in the family. There was no history of irritation or minor trauma. Dermatological examination revealed asymptomatic, rounded, yellowish to skin-coloured, measuring between 0.5 and 3 cm, smooth-surfaced, multiple coalescent papules, cerebriform plaques, and nodules. There was no tenderness, ulceration, excessive hair growth, pigmentation, comedo-like plugs, café au lait macules, or induration. Examination of the scalp and mucous membranes were normal. Routine laboratory tests were normal. No abnormality was found in systemic examination. Surgical excision was performed. Excision defect was repaired with transpositional flap. Aggregates of mature adipocytes were seen around the dermal blood vessels, eccrine glands, and between the collagen bundles. Histopathological examination was assessed as NLCS [Figure 2]. Patient healed uneventfully and aesthetic result was good [Figure 3]. No recurrence was seen in the 3-year follow-up.

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Figure 1

A 32-year-old male presented with multiple swelling over the sacrum

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Figure 2

Histopathologic examination of the NLCS is seen. Mature fat cells replace much of the dermis. Sometimes they extend to the undersurface of the epidermis (H and E, ×40 (left view), ×200 (right view))

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Figure 3

Early (above; 1 week) and late postoperative (below; 3 years) views of the operating site. Patient healed without any complication

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NLCS is a rare uncommon benign hamartomatous malformation.[12] Few cases of NLCS have been reported since it was first described by Hoffman and Zurhelle in 1921.[4] There is no sexual predilection or hereditary trend.[1] Our patient was a male with no history of similar lesions in the family. The classical form of NLCS is usually unilateral and may be band-like, linear, or zosteriform in distribution.[13] Our patient's lesions were located over the sacrum. Lesions were unusually extended both side of the midline. Surgical excision is a therapeutic modality for NLCS treatment. Recurrence of NLCS is rarely seen after surgical excision.[1–4] There are numerous surgical options to resurface the resultant cutaneous defect after excision of the lesion.[45] The simplest of these options consists of excision and direct closure of the defect.[5] However, if the defect cannot be closed by direct cutaneous advancement, other options for wound closure include local flaps. Our patient was treated with total surgical excision and repair of surgical defect with local flap without complication. Recurrence has not been seen for 3-year follow-up. The physician should be aware of this rare hamartomatous malformation which may be located on both sides of the midline over the sacrum. It can become large if left untreated.