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CASE REPORT
Year : 2001  |  Volume : 18  |  Issue : 1  |  Page : 66-69
 

Adrenal myelolipoma associated with cholelithiasis


Departments of Endocrine Surgery, Endocrine Medicine, Pathology and Radiology, Sanjay Gandhi-Postgraduate Institute of Medical Sciences, Lucknow., India

Correspondence Address:
Sushil K Gupta
Department of Endocrinology, SGPGIMS, Raebareli Road, Lucknow - 226 014
India
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Source of Support: None, Conflict of Interest: None


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   Abstract 

Myelolipomas are usually as ymptomatic and discovered incidentally or at autopsy. They are rare benign adrenal neoplasms, characterized by the presence of adipose tissue and elements of haematopoietic tissue in various degrees of differentiation. Occasionally these tumours may cause local symptoms or rupture. Not much literature is available on the association of gallstones with adrenal myelolipoma. It could be an incidental association or there may be a common underlying pathologic basis. We report clinical, radiologic and therapeutic aspects of a large myelolipoma of the right adrenal gland associated with gallstones in a patient with type 2 diabetes mellitus and diabetic nephropathy.


Keywords: Myelolipoma; Incidentiloma.


How to cite this article:
Kar DK, Agarwal A, Mishra AK, Gupta SK, Prasad KK, Sawlani V, Pal L, Mishra SK. Adrenal myelolipoma associated with cholelithiasis. Indian J Urol 2001;18:66-9

How to cite this URL:
Kar DK, Agarwal A, Mishra AK, Gupta SK, Prasad KK, Sawlani V, Pal L, Mishra SK. Adrenal myelolipoma associated with cholelithiasis. Indian J Urol [serial online] 2001 [cited 2020 May 27];18:66-9. Available from: http://www.indianjurol.com/text.asp?2001/18/1/66/37422



   Introduction Top


Myelolipoma of the adrenal gland is a rare benign cortical and hormonally inactive neoplasm. It is characterized by the presence of both adipose and haematopoietic tissues. Majority of adrenal myelolipoma are discovered incidentally (adrenal 'incidentalomas'). Two-thirds of cases are asymptomatic; however, larger masses may cause local symptoms.[1] Association of adrenal myelolipoma with cholelithiasis has not been widely described except for one case report.[2] We. hereby, report a case of the right adrenal gland myelolipoma presenting with gallstone dyspepsia in a subject with type 2 diabetes mellitus and diabetic nephropathy.


   Case Report Top


A 47-year-old male, known case of type 2 diabetes mellitus with hypertension, diabetic nephropathy with moderate chronic renal failure (creatinine 2.7 mg/dL), background diabetic retinopathy. autonomic neuropathy and sensorimotor peripheral neuropathy was referred to us for evaluation of large right adrenal mass and cholelithiasis detected on routine abdominal ultrasound for dyspepsia. He had good glycemic control with insulin and well-controlled blood pressure on drugs. One month prior to admission he required short-term hemodialysis for fluid overload and progressiveazotemia. Examination revealed pulse 116/min, BP 170/90 mm Hg with postural hypotension of 30 mm Hg and bilateral pitting pedal edema. Abdominal examination did not reveal any abdominal lump or organomegaly. Biochemical investigation revealed Hb. of 8.9 gm/dl, serum creatinine 3.6 mg/dL, Na/K 134/5.8 mEq/L. Other investigations were within normal limits.

Endocrine evaluation of the adrenal mass revealed normal 24 hours urinary metanephrine (1.38 mg/24 hours: normal <0.9 mg/24 hrs) and serum cortisol (overnight Dexamethasone Suppression Test: < 5pmg/dl). Plain and contrast enhanced CT scan abdomen showed evidence of a well defined, rounded right adrenal mass (5 x 7.4 cm) with homogenously non-enhancing low density tissue (-83 HF units) and a focal calcified area in the periphery [Figure - 1]. Left adrenal gland was normal. A gallstone was present in the neck of the gall bladder [Figure - 2]. This CT finding and hormonal evaluation suggested a diagnosis of nonfunctioning adrenal myelolipoma. In view of large size. a decision to surgically remove adrenomyelolipoma along with gall bladder in same sitting was undertaken. Transperitoneal exploration revealed single right adrenal mass measuring 6 x 5 x 3 cm without any adhesions/invasions to the other abdominal viscera and vessels. Right adrenalectomy with cholecystectomy was performed. Histopathology of adrenal mass revealed partial replacement of the parenchyma by lobules of mature fat cells along with haematopoietic cells [Figure - 3]. The sections from the gall bladder showed patchy denudation of mucosa, Rokintasky Aschoff sinus formation and mild mononuclear cell infiltrate. The postoperative recovery was uneventful.


   Discussion Top


Myelolipoma is an uncommon adrenal benign tumour with an incidence rate from 0.08% to 0.2% in autopsy series. [3] The exact aetiopathogenesis of these tumours is still obscure. Various hypotheses proposed are bone marrow embolism, extramedullary hematopoiesis, and bone marrow metaplasia. The most plausible explanation may be that undifferentiated mesenchymal stem cells within the adrenal cortex are stimulated to differentiate into myeloid and lipoid lines by some stress in the form of chronic illness, necrosis or neoplasia.[4] The review of the literature shows the relative frequency of myelolipomas as well as other benign adrenal tumours in patients with excessive secretion of ACTH.[5] Several authors reported an association between myelolipoma, hypertension and obesity.[6] Hypertension may be caused by compression of the renal artery or other mechanisms. The Doppler ultrasound study in our case could not document any renal artery involvement.

Adrenal myelolipomas are identified by their characteristic echogenicity of the adipose tissue in sonography. But one should keep in mind the differential diagnosis with angiomyolipoma of kidney or adrenal nonfunctioning neoplasm because of similar picture. CT is very sensitive and reveals characteristic picture, as it was observed in our case. In majority, it is possible to make a CT diagnosis of myelolipoma confidently. However, myeloid tissue, hemorrhage or calcium deposits may focally or diffusely increase the tumour attenuation values and the fat content may not be observed when they are extensive. Under these conditions differentiation from other adrenal neoplasms is difficult and may be indistinguishable. In such a situation, CT-guided FNAC biopsy can be obtained to make specific histologic diagnosis. In our case we did not obtain an FNAC because imaging findings were highly suggestive of myelolipoma. Further, a tissue diagnosis may be essential in giant myelolipomas, since radiologically, well-differentiated liposarcoma may mimic giant myelolipomas on CT.[7] Most cases of myelolipoma are asymptomatic and discovered incidentally; symptoms are reported as a consequence of haemorrhage, rupture or compression of the neighbouring structures: abdominal pain, dyspepsia resulting from compression of duodenum[8], dyspnoea, haematuria and nephrotic syndrome. Endocrine dysfunction has been reported in only 10% of cases and may take the form of Addison's disease, Cushing's disease, hermaphroditism, virilism, extreme obesity, or multiple endocrinopathy.[9]

Establishing the nature of incidentally discovered adrenal masses has been a major concern, because once they are discovered, their nature must be defined in order to exclude a metastatic lesion and to a lesser degree, a pheochromocytorna or a primary adrenal carcinoma. Another interesting finding in our patient was of associated gall stones. Literature search reveals only one case report of adrenolipoma occurring in association with cholelithiasis.2 Because it was a large tumour of size more than 6 cm and associated gall stone in a patient of NIDDM, surgical treatment is justified to prevent future risk of spontaneous haemorrhage and gall stone associated complications in a patient of NIDDM. Despite the fact that adiposity is associated with adrenolipoma as well as gall stones, the occurrence of adrenolipoma and gall stones is rarely reported. The reason of this is unknown. The co-occurrence of these two pathologies may be an incidental finding. However, it would be interesting to look for a common~pathophysiologic basis because myelolipomas are known to be associated with obesity. Further prospective studies of co-occurrence of adrenolipoma and cholelithiasis are required.

 
   References Top

1.Kazan E. Engel IA. Zirinsky K et al. Sectional imaging of the adrenal glands, computed tomography and ultrasound. In: Daracott Vaughan Jr E, Carey RM (eds.). Adrenal disorders. Thiem Med Publ. New York. 1989; 37-38.  Back to cited text no. 1    
2.Solis Daun JO, Rodriguez Garcia R. Torres Trejo A, Alor Gunman M. The adrenal myelolipoma associated with lithiasic cholecystitis, hiatal hernia and esophagitis. Rev Gastroenterol Mex 1999; 64: 143-145.  Back to cited text no. 2    
3.Vick CW, Zema RK, Marines E, Cronan, Walsh JW. Adrenal myelolipoma: CT and ultrasound findings. Urol Radiol 1984; 6: 7-13.  Back to cited text no. 3    
4.Selye H. Stone H. Hormonally induced transformation of adrenal into adipose tissue. Am J Pathol 1950: 26: 211-233.  Back to cited text no. 4    
5.Ravichandran R, Lafferty F, McGinnis MJ, Taylor HC. Congenital adrenal hyperplasia presenting as massive adrenal incidentalomas in the sixth decade of life: report of two patients with 21-hydroxylase deficiency. J Clin Endocrinol Metab 1996; 81: 1776-1779.  Back to cited text no. 5    
6.Diekrnann KP. Hamm B, Pickartz H, Jonas D, Bauer HW. Adrenal myelolipoma: Clinical, radiologic and histologic features. Urology 1987: 29: 1-8.  Back to cited text no. 6    
7.Dunphy CH. Computed Tomography-Guided Fine Needle Aspiration Biopsy of Adrenal Myelolipoma: Case Report and Review of Literature. Acta Cytologica 1991: 35: 353-356.  Back to cited text no. 7    
8.Muraro GB. Melone F, Dami A, Farina U, Colagrande S. Myelolipoma of the adrenal gland presenting with dyspepsia and epigastric pain. Urol Int 1998; 52: 217-219.  Back to cited text no. 8    
9.Plant A. Myelolipoma in the adrenal cortex (myeloadipose structures). Am J Pathol 1958; 34: 487.   Back to cited text no. 9    


    Figures

  [Figure - 1], [Figure - 2], [Figure - 3]



 

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    Abstract
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