Peri-portal lymphedema in association with an acute adrenal insufficiency: case report

Journal of Medical Case Reports

Fatheya Alawadi²  Sona Abuelkheir²  Azza Abdulaziz²  Inas A Al Hameedi¹  Alaaeldin MK Bashier²  Elamin Ibrahim Elamin Abdelgadir² 

[1] Radiology Department, Dubai Hospital, Alkhaleej road, Dubai, P.O.BOX: 7272, United Arab Emirates;Endocrine Unit, Dubai Hospital, Alkhaleej road, Dubai, P.O.BOX: 7272, United Arab Emirates

关键词: Steroids;    Peri-portal lymphedema;    Lymphedema;    Free water excretion;    Crisis;    Adrenal insufficiency;    Addison’s disease;   

Others  :  822495
DOI  :  10.1186/1752-1947-8-98

 received in 2013-07-30, accepted in 2014-02-18,  发布年份 2014

【 摘 要 】

Introduction

We report the case of a patient with peri-portal lymphedema in association with severe adrenal insufficiency. To the best of our knowledge, this association was not previously reported in the literature. Peri-portal lymphedema is usually seen in cases such as blunt abdominal trauma, hepatic congestion and post–liver transplantation.

Case presentation

We present the case of a 28-year-old Indian man who presented to our hospital with adrenal crisis and was treated accordingly. Computed tomography of his abdomen showed evidence of peri-portal lymphedema (edema) with some free fluid collection. We excluded other causes of this pathology and followed the patient’s condition after steroid replacement therapy. We found no other contributing factors to the patient’s peri-portal lymphedema apart from the adrenal crisis, which was more consolidated when we followed the patient after steroid replacement therapy, during which follow-up computed tomography showed complete resolution of the pathology.

Conclusions

We conclude following an extensive MEDLINE® search that this is the first case to be reported for the association between peri-portal lymphedema and adrenal insufficiency, after having excluded all other causes of peri-portal lymphedema. This signifies reporting of this case as the first one in the medical literature.

【 授权许可】

   
2014 Abdelgadir et al.; licensee BioMed Central Ltd.

【 图 表 】

Figure 1.

Figure 2.

【 参考文献 】

• [1]Aggarwal A, Kotru M, Sharma V, Sharma S: Adrenal insufficiency in primary adrenal lymphoma: innocuous presentation of a rare sinister illness. Niger J Clin Pract 2011, 14:115-117.
• [2]Mota FC, Matos C, Marques O: Primary adrenal insufficiency: from clinical presentation to genetic counselling [Abstract]. Bristol, UK: Society for Endocrinology Clinical Update 2007; 2007. Available at http://www.bioscilibrary.com/resource/clinicalupdate/cu07/Primary%20Adrenal%20Insufficiency%20from%20clinical%20presentation%20to%20genetic%20counseling.pdf webcite (accessed 26 February 2014)
• [3]Myers KA, Kline GA: Addison disease presenting with acute neurologic deterioration: a rare presentation yields new lessons from old observations in primary adrenal failure. Endocr Pract 2010, 16:433-436.
• [4]Roldan-Martin MB, Rodriguez-Ogando A, Sanchez-Galindo AC, Parente-Hernandez A, Luengo-Herrero V, Sanchez-Sanchez C: Rare presentation of shock and acute mesenteric ischaemia secondary to acute adrenal insufficiency in an 11-year-old male. J Paediatr Child Health 2013, 49:498-500.
• [5]Kunjal RA, Ramadoo RR, Teelucksingh S, Naraynsingh V: Adrenal insufficiency and bowel obstruction: an overlooked association. Case Rep Clin Med 2013, 2:111-114.
• [6]Boulton R, Hamilton MI, Dhillon AP, Kinloch JD, Burroughs AK: Subclinical Addison’s disease: a cause of persistent abnormalities in transaminase values. Gastroenterology 1995, 109:1324-1327.
• [7]Rizvi AA, Kerrick JG: Liver involvement and abnormal variables in undiagnosed Addison’s disease. Endocr Pract 2001, 7:184-188.
• [8]Olsson RG, Lindgren A, Zettergren L: Liver Involvement in Addison’s disease. Am J Gastroenterol 1990, 85:435-438.
• [9]O’Beirne J, Holmes M, Agarwal B, Bouloux P, Shaw S, Patch D, Burroughs A: Adrenal insufficiency in liver disease—What is the evidence? J Hepatol 2007, 47:418-423.
• [10]Ly JN, Miller FH: Periportal contrast enhancement and abnormal signal intensity on state-of-the-art MR images. AJR Am J Roentgenol 2001, 176:891-897.
• [11]Lee SH, Han YM, Choi KC, Kim CS, Lee SY, Chung GH, Sohn MH, Yang DH, Lee YW: [Periportal low-attenuation: a CT sign of lymphatic obstruction] [in Korean]. J Korean Radiol Soc 1995, 33:87-92. Korean
• [12]Patrick LE, Ball TI, Atkinson GO, Winn KJ: Paediatric blunt abdominal trauma: periportal tracking at CT. Radiology 1992, 183:689-691.
• [13]Koslin DB, Stanley RJ, Berland LL, Shin MS, Dalton SC: Hepatic perivascular lymphedema: CT appearance. AJR Am J Roentgenol 1988, 150:111-113.
• [14]Cutler RE, Kleeman CR, Koplowitz J, Maxwell MH, Dowling JT: Mechanisms of impaired water excretion in adrenal and pituitary insufficiency. III. The effect of extracellular or plasma volume expansion, or both, on impaired diuresis. J Clin Invest 1962, 41:1524-1530.
• [15]Kleeman CR, Czaczkes JWV, Cutler R: Mechanisms of impaired water excretion in adrenal and pituitary insufficiency. IV. Antidiuretic hormone in primary and secondary adrenal insufficiency. J Clin Invest 1964, 43:1641-1648.