Ami Gokli1, Jeremy Neuman2, Ruby Lukse1, June Koshy1, Fanyi Kong3, Tal Laor4. 1. Department of Radiology, Staten Island University Hospital, 475 Seaview Ave., Staten Island, NY, 10305, USA. 2. Department of Radiology, Staten Island University Hospital, 475 Seaview Ave., Staten Island, NY, 10305, USA. jneuman@northwell.edu. 3. Department of Pathology, Staten Island University Hospital, Staten Island, NY, USA. 4. Department of Radiology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.
Abstract
BACKGROUND: Childhood asymmetrical labium majus enlargement (CALME) has been described sparsely in recent surgery, pathology, pediatric and gynecology literature; however, no comprehensive description from a radiology perspective has been developed. OBJECTIVE: The purpose of this case series is to describe the imaging findings of CALME and to review the current understanding of this recently described clinical entity with regard to clinical presentation, pathophysiology, differential diagnosis and treatment options. MATERIALS AND METHODS: This is a retrospective analysis of 3 girls, ages 5-7 years, who presented for imaging evaluation with subsequent pathologically proven CALME. Each child's clinical history, length of symptoms, imaging appearance and pathological findings were reviewed. RESULTS: All three girls presented with unilateral enlargement of the labium majus (two right-side, one left-side) with no history of trauma or other inciting cause. Two girls had painless labial enlargement that was recognized for weeks, and one had similar symptoms for 1 year prior to presentation. One girl was evaluated initially with sonography, and all three children underwent MR imaging. Sonographic evaluation showed asymmetrical labial enlargement without a definable mass. In each girl, the MR imaging findings were characterized by relatively ill-defined T1-weighted hypointense signal, T2-weighted hypo- to isointense signal with interspersed hyperintense septae, and heterogeneous patchy and feathery strands of enhancement on post-contrast imaging. Biopsy from each child showed benign fibrous tissue with intervening mature fibroadipose tissue, vessels and nerves without findings of inflammation or neoplasia. CONCLUSION: The MR imaging appearance of CALME is consistent. Recognition and appreciation of this unique pediatric entity by the radiologist may be essential for appropriate diagnosis and can help to guide therapy. Current preferred treatment approach is conservative.
BACKGROUND: Childhood asymmetrical labium majus enlargement (CALME) has been described sparsely in recent surgery, pathology, pediatric and gynecology literature; however, no comprehensive description from a radiology perspective has been developed. OBJECTIVE: The purpose of this case series is to describe the imaging findings of CALME and to review the current understanding of this recently described clinical entity with regard to clinical presentation, pathophysiology, differential diagnosis and treatment options. MATERIALS AND METHODS: This is a retrospective analysis of 3 girls, ages 5-7 years, who presented for imaging evaluation with subsequent pathologically proven CALME. Each child's clinical history, length of symptoms, imaging appearance and pathological findings were reviewed. RESULTS: All three girls presented with unilateral enlargement of the labium majus (two right-side, one left-side) with no history of trauma or other inciting cause. Two girls had painless labial enlargement that was recognized for weeks, and one had similar symptoms for 1 year prior to presentation. One girl was evaluated initially with sonography, and all three children underwent MR imaging. Sonographic evaluation showed asymmetrical labial enlargement without a definable mass. In each girl, the MR imaging findings were characterized by relatively ill-defined T1-weighted hypointense signal, T2-weighted hypo- to isointense signal with interspersed hyperintense septae, and heterogeneous patchy and feathery strands of enhancement on post-contrast imaging. Biopsy from each child showed benign fibrous tissue with intervening mature fibroadipose tissue, vessels and nerves without findings of inflammation or neoplasia. CONCLUSION: The MR imaging appearance of CALME is consistent. Recognition and appreciation of this unique pediatric entity by the radiologist may be essential for appropriate diagnosis and can help to guide therapy. Current preferred treatment approach is conservative.
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