Giant Nevus Lipomatosus Cutaneous Superficialis with Intramuscular Lipomatosis Caused Sciatic Nerve Compression — Suleyman Tas PubMed

LETTER TO THE EDITOR Giant Nevus Lipomatosus Cutaneous Superficialis with Intramuscular Lipomatosis Caused Sciatic Nerve Compression To the Editor: Nevus lipomatosus cutaneous superficialis (NLCS) is a very rare disorder that Hoffman and Zuhrelle first described in 1921. 1 It is characterized by accumulation of mature adipose tissue in the dermis. 1 These lesions are usually congenital and remain asymptomatic for the duration of their existence. 2 We encountered a giant NLCS with intramuscular lipomatosis that caused sciatic nerve compression. A 45-year-old man was admitted with a slowly growing, huge mass on the sacral area for 15 years’ duration and numbness in the left leg for 1 month. A physical examination revealed a 50 3 50 cm rubbery, nontender, soft, verrucous tumor (Figure 1A). There were neither cafe´-au-lait spots nor developmental defects. He had complete excision of a 10 3 5 cm tumor on the sacral area 5 years before at an external center. Magnetic resonance imaging (MRI) performed before surgery demonstrated diffuse intramuscular lipomatosis associated with a giant NLCS between the gluteal muscles that extended to the thigh region and compressed the sciatic nerve (Figure 2). MRI measurements of the intramuscular lipoma with an association with NLCS were 503 20 3 10 cm. The patient’s medical history and the results of routine laboratory testing were unremarkable. A wide excision was planned due to sciatic nerve compression and discomfort in the patient’s daily life. After wide surgical resection, the defect was closed with a large transposition flap and the donor site was repaired with a split-thickness skin graft. At the 24-month follow- up, there was no recurrence and no symptom of compres- sion neuropathy (Figure 1B). The histopathologic findings demonstrated fat infiltration from the upper dermis into the deep muscle layers and indicated NLCS with intramuscular lipomatosis (Figure 3). Treatment was deemed unnecessary as NLCS remains asymptomatic; other than cosmesis, simple excision is considered sufficient.2 There is only one reported sympto- matic case of NLCS with compression neuropathy of the ulnar nerve. 2 In this case, the authors performed partial excision and suggested partial excision because of the increased morbidity of total excision. Again, there is only one case of a giant NLCS that describes fat infiltration extension to the deep layer of muscle. 3 In this report, although complete surgical excision was performed, NLCS recurred and reached a huge size. 3 We suggest that MRI be done for detecting any muscular component of NLCS for curative treatment in giant NLCS. To the best of our knowledge, there are only six cases of giant NLCS (over 15 3 15 cm) in the literature, including this report, and two of them have muscular lipomatosis under NLCS. Physicians should be well aware of this very rare condition because early recognition DOI 10.2310/7750.2013.13161 # 2014 Canadian Dermatology Association Figure 1. A, A huge (50 3 50 cm), soft, nontender, rubbery, cerebriform mass in the sacral area and a sub- dermal excessive mass extend from the gluteal region to the thigh. B, The examination at postoperative 24 months showed no recurrence. Canadian Dermatology Association | Journal of Cutaneous Medicine and Surgery, Vol 18, No 4 (July/August), 2014: pp 221–222 221

provides for more conservative resection and less invasive reconstruction. Even though NLCS is described as a fat metaplasia in connective tissue, its behavior is more aggressive if it has an intramuscular component. Thus, in these cases, wide surgical excision should be planned for preventing recurrent and giant NLCS. However, the report presented here is the largest NLCS and is the first NLCS that caused sciatic nerve compression. Acknowledgment Financial disclosure of authors and reviewers: The authors declare that they have no conflicts of interest, commercial associations, or intent of financial gain regarding this research. Su¨leyman Tas¸ Hu¨samettin Top Department of Plastic, Reconstructive and Aesthetic Surgery Trakya University, Faculty of Medicine Edirne, Turkey References 1. Hoffman E, Zurhelle E. Uberneinen Naevus lipomatodes cutaneus superficialis der linken Glutaagegend. Arch Dermatol Syphilol 1921; 130:327–33. 2 .T u n c eS ,S e z g i nB ,Y i l m a zG ,e ta l .C o m p r e s s i o nn e u r o p a t h yc a u s e d by an unusual lesion: nevus lipoma tosus cutaneus superficialis. Plast Reconstr Surg 2011;127:72–4, doi:10.1097/PRS.0b013e31820633fb. 3. Jung ST, Park HW, Yun SJ. Giant nevus lipomatosus cutaneous superficialis with intramuscular lipomatosis. J Am Acad Dermatol 2012;67:168–70, doi:10.1016/j.jaad.2012.01.025. Figure 2. Magnetic resonance ima ge showing a giant nevus lipomatosus cutaneous superficialis infiltrating as an intramuscular lipomatosis with expansion of the gluteus major muscle and displacement of the sciatic nerve posteriorly. Figure 3. A, Mature fatty tissue between collagen bundles of the reticular dermis, suggestive of nevus lipomatosus cuta- neous superficialis. B,M a t u r ea d i p o s e tissue infiltrating and encasing the mus- cle (hematoxylin–eosin stain;350 origi- nal magnification). Letter to the Editor 222 Canadian Dermatology Association | Journal of Cutaneous Medicine and Surgery, Vol 18, No 4 (July/August), 2014: pp 221–222