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Adrenal lymphangioma: clinicopathologic and immunohistochemical characteristics of a rare lesion.
Hum Pathol. 2011 Jul; 42(7):1013-8.HP

Abstract

Adrenal lymphangiomas, also known as cystic adrenal lymphangiomas, are rare, benign vascular lesions that usually remain asymptomatic throughout life. Although previously adrenal lymphangioma lesions were primarily found at autopsy, they are currently detected during imaging work-up for unrelated causes and are likely to imitate other adrenocortical or adrenal medullary neoplasms. We aimed to retrospectively review all adrenal lymphangioma cases at our hospital and further document their lymphatic origin by immunohistochemical staining. A search of surgical pathology records (1984-2008) was conducted. All hematoxylin and eosin sections were retrieved from archives and reviewed by 2 pathologists in the study. Clinical information was gathered from electronic medical records. Representative paraffin-embedded sections from each case were selected for immunohistochemical analysis using monoclonal antibodies D2-40 and AE1/AE3. A total of 9 adrenal lymphangioma cases were identified (6 women and 3 men). All 9 patients were adults at time of diagnosis with a mean age of 42 years (range, 28-56 years). There were 7 white patients, 1 African American patient, and 1 Asian patient. The average size of an adrenal lymphangioma lesion was 4.9 cm (range, 2.0-13.5 cm). Adrenal lymphangioma was twice more frequently located on the right side (6 right-sided and 3 left-sided). Clinically, 4 (44%) of the 9 lesions presented with abdominal, flank, or back pain. One lymphangioma was found during work-up for labile hypertension. The remaining 4 lesions (44%) were asymptomatic and incidentally found during imaging studies for unrelated causes. Surgical removal was achieved by total adrenalectomy in 8 of the 9 lesions and by partial adrenalectomy in the remaining case. No evidence of recurrence or development of a contralateral lesion was encountered in any of the patients. Histologically, our adrenal lymphangiomas showed a typical multicystic architecture with dilated spaces lined by flattened, bland, simple lining. The cystic channels/spaces occasionally contained proteinaceous material and lacked red blood cell content. On immunohistochemical stains, D2-40 cytoplasmic staining was positive in all 9 examined lesions, whereas AE1/AE3 was negative, thus, confirming their lymphatic nature. D2-40 staining was diffuse in 2 and focal in the 7 remaining lesions. Adrenal lymphangiomas are very rare, benign lymphatic neoplasms with a female, right-sided predominance in our current series. They may clinically present with abdominal pain or can be incidentally found during adulthood as a mass, necessitating surgical removal to rule out other types of adrenal neoplasms.

Authors+Show Affiliations

Department of Pathology, The Johns Hopkins Hospital, Baltimore, MD 21287, USA.No affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article

Language

eng

PubMed ID

21315417

Citation

Ellis, Carla L., et al. "Adrenal Lymphangioma: Clinicopathologic and Immunohistochemical Characteristics of a Rare Lesion." Human Pathology, vol. 42, no. 7, 2011, pp. 1013-8.
Ellis CL, Banerjee P, Carney E, et al. Adrenal lymphangioma: clinicopathologic and immunohistochemical characteristics of a rare lesion. Hum Pathol. 2011;42(7):1013-8.
Ellis, C. L., Banerjee, P., Carney, E., Sharma, R., & Netto, G. J. (2011). Adrenal lymphangioma: clinicopathologic and immunohistochemical characteristics of a rare lesion. Human Pathology, 42(7), 1013-8. https://doi.org/10.1016/j.humpath.2010.10.023
Ellis CL, et al. Adrenal Lymphangioma: Clinicopathologic and Immunohistochemical Characteristics of a Rare Lesion. Hum Pathol. 2011;42(7):1013-8. PubMed PMID: 21315417.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Adrenal lymphangioma: clinicopathologic and immunohistochemical characteristics of a rare lesion. AU - Ellis,Carla L, AU - Banerjee,Priya, AU - Carney,Erin, AU - Sharma,Rajni, AU - Netto,George J, Y1 - 2011/02/11/ PY - 2010/08/31/received PY - 2010/10/18/revised PY - 2010/10/20/accepted PY - 2011/2/15/entrez PY - 2011/2/15/pubmed PY - 2011/9/15/medline SP - 1013 EP - 8 JF - Human pathology JO - Hum. Pathol. VL - 42 IS - 7 N2 - Adrenal lymphangiomas, also known as cystic adrenal lymphangiomas, are rare, benign vascular lesions that usually remain asymptomatic throughout life. Although previously adrenal lymphangioma lesions were primarily found at autopsy, they are currently detected during imaging work-up for unrelated causes and are likely to imitate other adrenocortical or adrenal medullary neoplasms. We aimed to retrospectively review all adrenal lymphangioma cases at our hospital and further document their lymphatic origin by immunohistochemical staining. A search of surgical pathology records (1984-2008) was conducted. All hematoxylin and eosin sections were retrieved from archives and reviewed by 2 pathologists in the study. Clinical information was gathered from electronic medical records. Representative paraffin-embedded sections from each case were selected for immunohistochemical analysis using monoclonal antibodies D2-40 and AE1/AE3. A total of 9 adrenal lymphangioma cases were identified (6 women and 3 men). All 9 patients were adults at time of diagnosis with a mean age of 42 years (range, 28-56 years). There were 7 white patients, 1 African American patient, and 1 Asian patient. The average size of an adrenal lymphangioma lesion was 4.9 cm (range, 2.0-13.5 cm). Adrenal lymphangioma was twice more frequently located on the right side (6 right-sided and 3 left-sided). Clinically, 4 (44%) of the 9 lesions presented with abdominal, flank, or back pain. One lymphangioma was found during work-up for labile hypertension. The remaining 4 lesions (44%) were asymptomatic and incidentally found during imaging studies for unrelated causes. Surgical removal was achieved by total adrenalectomy in 8 of the 9 lesions and by partial adrenalectomy in the remaining case. No evidence of recurrence or development of a contralateral lesion was encountered in any of the patients. Histologically, our adrenal lymphangiomas showed a typical multicystic architecture with dilated spaces lined by flattened, bland, simple lining. The cystic channels/spaces occasionally contained proteinaceous material and lacked red blood cell content. On immunohistochemical stains, D2-40 cytoplasmic staining was positive in all 9 examined lesions, whereas AE1/AE3 was negative, thus, confirming their lymphatic nature. D2-40 staining was diffuse in 2 and focal in the 7 remaining lesions. Adrenal lymphangiomas are very rare, benign lymphatic neoplasms with a female, right-sided predominance in our current series. They may clinically present with abdominal pain or can be incidentally found during adulthood as a mass, necessitating surgical removal to rule out other types of adrenal neoplasms. SN - 1532-8392 UR - https://www.unboundmedicine.com/medline/citation/21315417/Adrenal_lymphangioma:_clinicopathologic_and_immunohistochemical_characteristics_of_a_rare_lesion_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S0046-8177(10)00415-6 DB - PRIME DP - Unbound Medicine ER -