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At least 19 recordsLinked to original sources
PubMed2006

Adrenal myelolipomas show nonrandom X-chromosome inactivation in hematopoietic elements and fat: support for a clonal origin of myelolipomas.

Myelolipomas are defined as mature fat associated with hematopoietic elements, often found in the adrenal gland. The question of whether the hematopoietic cells are truly "normal" has not been evaluated extensively. In this study, we evaluated histologic, immunohistochemical features and comparisons of X-chromosome inactivation patterns in 19 myelolipomas. Formalin-fixed, paraffin-embedded tissue from 19 myelolipomas was stained with hematoxylin and eosin and immunostained with monoclonal antibodies against CD138, CD34, CD117, CD42a, hemoglobin, myeloperoxidase, collagen IV, and nerve growth factor receptor. Histologic evaluation included estimates of overall cellularity of hematopoietic tissue, estimates of cellularity in the areas of highest concentration of hematopoietic tissue, myeloid to erythroid ratio, and numbers of megakaryocytes. X-chromosome inactivation analysis was performed on myelolipomas from 11 female patients by polymerase chain reaction. Myelolipomas showed wide variation in cellularity within the lesion (5% to 90%) with no correlation with the patient's age. All the myelolipomas demonstrated normal trilineage hematopoiesis and cellular morphology, with few early myeloid precursors, as evidenced by negativity for CD117 and only rare positivity for CD34 antibodies. Most of the myelolipomas (14/18) had markedly increased megakaryocytes compared with normal marrows. The majority of myelolipomas also had a stromal composition and vascular patterns that were different from those of normal bone marrow. X-chromosome inactivation studies demonstrated nonrandom X-chromosome inactivation in 8/11 myelolipomas from female patients. Myelolipomas are morphologically different from the normal bone marrow. The majority of myelolipomas also have nonrandom X-chromosome inactivation, suggesting a clonal origin for these tumors.

Adipose Tissue↗
PubMed1982

Extra-adrenal myelolipomas compared with extramedullary hematopoietic tumors: a case of presacral myelolipoma.

A large presacral myelolipoma associated with increasing constipation is reported in a 70-year-old obese diabetic female. The resected tumor was encapsulated and dark red to yellow. Light and electron microscopy revealed mature and immature hematopoietic tissue, lymphoid aggregates and mature adipocytes. Myelolipomas arising in extra-adrenal sites are rare. Considered in the differential diagnosis are reactive extramedullary hematopoietic tumors that usually occur in patients with severe chronic anemias. Patients with extramedullary hematopoietic tumors are characterized by anemia, frequent hepatosplenomegaly and abnormal peripheral blood smears, and may be any age. The tumors are usually multiple and usually located in the mediastinum or epidural space. In contrast, patients with extra-adrenal myelolipomas are usually older than 40 years, have normal blood studies, absent hepatosplenomegaly, and usually have chronic debilitating diseases or endocrinopathies. Extra-adrenal myelolipomas are single and usually occur within the abdomen. Extramedullary hematopoietic tumors have a predominance of the hematopoietic component, with erythroid hyperplasia of that component. Extra-adrenal myelolipomas may have a predominance of either the hematopoietic or fatty component, chiefly the latter, and generally have a more conspicuous lymphocyte population. Although the components of these tumors are the same, we consider them separable clinically, pathogenetically and, in many cases, pathologically.

Aged↗
PubMedDate not supplied

Imaging and pathologic features of myelolipoma.

Myelolipoma is a benign tumor consisting of mature fat interspersed with hematopoietic elements resembling bone marrow. Imaging findings in a large series of pathologically proved cases of myelolipoma were correlated with the pathologic and histologic features of the lesions. Myelolipoma manifests in four distinct clinicopathologic patterns: isolated adrenal myelolipoma, adrenal myelolipoma with hemorrhage, extraadrenal myelolipoma, and myelolipoma associated with other adrenal disease. Myelolipoma is difficult or impossible to detect at plain radiography unless the lesion is large and predominantly fatty. At ultrasound, myelolipoma often has heterogeneous echogenicity due to its typically nonuniform architecture. Computed tomography (CT) frequently demonstrates large amounts of fat with areas of interspersed higher-attenuation tissue. At magnetic resonance imaging, predominantly fatty areas usually have increased signal intensity on T1-weighted images and moderate hyperintensity complicated by the presence of marrowlike elements in the corresponding regions on T2-weighted images. The imaging appearance of myelolipoma is altered by the presence of hemorrhage. In such cases, CT is the most accurate method for evaluation. Knowledge of the imaging characteristics of myelolipoma usually allows presumptive diagnosis, although percutaneous needle biopsy may be needed to confirm the diagnosis in cases of extraadrenal myelolipoma. Surgical excision is unnecessary unless the diagnosis is unclear or the lesion is symptomatic. Asymptomatic, nonhemorrhagic myelolipomas do not require therapy.

Adrenal Gland Diseases↗
PubMed1998

Myelolipoma: CT and pathologic features.

PURPOSE: To correlate clinical and pathologic patterns of myelolipoma with the computed tomographic (CT) appearance. MATERIALS AND METHODS: All cases of myelolipoma accessioned by the Armed Forces Institute of Pathology from 1981 through 1997 were reviewed. CT images were scored for the location and size of each myelolipoma and the presence of calcification, hemorrhage, fat, and pseudocapsule. Pathologic findings for the pattern of fat and bone marrow elements were correlated with CT findings. RESULTS: In 74 patients, 86 myelolipomas were found, of which 72 were in an adrenal gland (eight were bilateral), and 14 extra-adrenal masses were found in 10 patients. Four clinicopathologic patterns emerged: (a) isolated adrenal myelolipoma in 37 patients (fat evident at CT, no other disorders present); (b) myelolipoma with hemorrhage in nine patients (imaging features similar to those of isolated adrenal myelolipomas but larger [mean diameter, 14.2 vs 9.9 cm; P = .01]; (c) extra-adrenal myelolipoma in 10 patients (imaging findings similar to those of adrenal myelolipomas, found most often in the retroperitoneum); and (d) myelolipomatous foci within other adrenal pathologic conditions in 18 patients (smaller, lower fat content, more heavily calcified). CONCLUSION: Myelolipomas are adrenal or extra-adrenal masses, with hemorrhage more common in larger lesions (diameter, > 10 cm). The CT appearance of myelolipomatous foci, which can be found within other pathologic adrenal conditions, is different from that of isolated adrenal myelolipomas.

Adipose Tissue↗
PubMed2002

Incidentally discovered adrenal myelolipoma. Report on 3 operated patients and update of the literature.

INTRODUCTION AND OBJECTIVES: Adrenal myelolipomas are rare benign non functioning tumors which are mostly detected incidentally by imaging (8-15%). The aim of this study was to find out the clinical outcome of 3 patients operated for incidentally discovered adrenal myelolipoma as well as to update the literature concerning this topic. PATIENTS AND METHODS: During a 24 year period (1976-2000) we operated on 28 of 39 (72%) patients for incidentally discovered adrenal mass and pathology assessed 3 myelolipomas (11%). All patients were males and average age was 54.3 years (range 46-66). Myelolipoma was associated with arterial hypertension in 2 patients and BPH in 1. Patients underwent unilateral adrenalectomy. Results Myelolipomas, all unilateral, were right-sided in 2 cases (67%) and left sided in 1. Average tumor diameter, as detected by CT scan, resulted 10.3 cm range (3-17). Endocrine investigation was negative. Average tumor size, as detected by pathology, resulted 9.3 cm (range 3-14) and mean weight 653 gr (range 210-980). Histopathology assessed primary adrenal myelolipoma in all cases. Mean follow-up is 110.6 months (range 102-128) and all patients are alive and disease free. CONCLUSIONS: Ultrasound (US), computed tomography (CT) and magnetic resonance imaging (MRI) are effective in diagnosing adrenal myelolipomas in about 90% of cases. Fine needle aspiration (FNA) cytology under CT or US control has an important role in investigating as well as diagnosing undefined masses suspected of adrenal or extraadrenal myelolipoma thus avoiding unnecessary surgery. The increasing number of incidentally discovered adrenal myelolipomas arise the question of appropriate treatment modalities which range from watchful waiting to surgical removal. Small asymptomatic tumors sized less than 4 cm can be monitored expectantly. Tumors when symptomatic or larger than 4 cm should be removed since the risk of spontaneous rupture with retroperitoneal hemorrhage and life threatening shock. Bilateral adrenalectomy for large and symptomatic tumors implies lifelong substitution with hydrocortisone. Actually, laparoscopic adrenalectomy is the new gold standard in treating myelolipomas since both showing more effective postoperative recovery than open surgery as well as reducing the number of patients who undergo long and close follow up.

Adrenal Gland Neoplasms↗
PubMed2006

[Giant bilateral adrenal myelolipoma associated with congenital adrenal hyperplasia].

BACKGROUND: Myelolipoma is a rare benign tumor formed by mature fat tissue with strata of haematopoiesis. It is mainly asymptomatic, only large myelolipomas manifest themselves by abdominal discomfort, bleeding or by symptoms of oppressing adjacent organs. It is usually localized in the region of the adrenal gland. Myelolipomas are mostly clinically inert, only a small number of them are associated with Cushing's type of endocrine disorders, Conn's syndrome, Addison's disease, etc. PATIENT AND METHODS: The authors present a rare case of a giant bilateral myelolipoma emerging out of the adrenal gland cortex in a congenital adrenal hyperplasia, with steroid 21-hydroxylase deficiency, in a woman with pronounced virilism. The principal diagnostic methods include sonography and computer tomography. The therapy available is either a surgical extirpation in the case of large or growing myelolipomas or observation in the case of small ones (< 5 cm). RESULTS: The condition was resolved by bilateral adrenalectomy with extirpation of both myelolipomas and subsequent hormonal substitution treatment during which there was a gradual regression of virilising symptoms. CONCLUSIONS: The coincidence of myelolipoma and congenital disorder with subsequent overproduction of the adrenocorticotropin hormone and androgens, might be explained by the incipient of myelolipoma through chronic hormonal stimulation of the adrenal gland cortex. However, the etiopathogenesis of myelolipoma is still unclear.

Adrenal Gland Neoplasms↗
PubMed2004

Case of combined adrenal cortical adenoma and myelolipoma.

We report a case of myelolipoma 10 mm in size within a functional cortical adenoma that was 33 x 22 x 17 mm in size. A 29-year-old woman was referred to hospital for transient hypertension. A right adrenal tumor was detected by computed tomography (CT) scan and magnetic resonance imaging (MRI). Her cortisol levels indicated a loss of the normal diurnal pattern, and urinary 17-hydroxycorticosteroids was elevated. She underwent a right adrenalectomy under the diagnosis of adrenal adenoma with Cushing's syndrome. The tumor was fairly well encapsulated by a thin layer of connective tissue. The major tumor portion was composed of two distinct cell types, clear cells and eosinophilic cells, arranged in an alveolar structure. These findings were representative of cortical adenoma. The adrenal cortical adenoma centrally included well-demarcated adipose tissue admixed with scattered islands of myelopoietic elements: erythroblasts, myeloid cell series and lymphocytic cells, which was eventually recognized as myelolipoma. Recently, adrenal myelolipoma has commonly been found because of the ease of detecting it as an incidentaloma by CT scan or MRI. However, the present adrenal myelolipoma case is uncommon because it is combined with a functioning cortical adenoma. Only six similar cases have previously been reported in English and Japanese publications. Furthermore, in the present case, the myelolipoma formed a tumor nodule, and to our knowledge, this is the first reported case of a radiographically recognizable tumor nodule. We discuss the etiology of myelolipoma and suggest that myelolipoma can develop in the course of endocrine hyperfunction.

Adenoma↗
PubMed1990

[A case of adrenal myelolipoma associated with adrenogenital syndrome].

Myelolipoma is a benign non-functioning tumor, and the number of documented cases is increasing in recent years. We report a case of adrenal myelolipoma associated with adrenogenital syndrome. A 65-year-old woman presented with a complaint of abdominal discomfort. CT showed a mass with abundant fatty density in the left adrenal gland suggesting myelolipoma. She was small and thin. External genitalia had a female appearance, but showed type III abnormality in Prader's classification with clitoral hypertrophy. She had a history of primary amenorrhea. Endocrinological examination revealed marked increase in the 17 alpha-hydroxyprogesterone (or 17-OHP) and pregnanetriol levels. On the basis of clinical features and laboratory values, simple virilizing type congenital adrenal hyperplasia due to 21-hydroxylase deficiency was made. The left adrenal tumor was surgically removed and histologically proved to be myelolipoma. Tumor-uninvolved adrenal area adjacent to myelolipoma showed adrenocortical hyperplasia. To our knowledge, this is the first reported case of adrenal myelolipoma to be associated with congenital adrenal hyperplasia, due to 21-hydroxylase deficiency in Japan. The etiology of myelolipoma remains to be not clarified but our case suggests involvement of endocrine disorder in the development of this tumor.

Adrenal Gland Neoplasms↗
PubMed2004

Combined adrenal adenoma and myelolipoma in a patient with Cushing's syndrome: case report and review of the literature.

Myelolipoma is an uncommon benign tumor of unknown etiology and adrenal myelolipoma is rarely associated with endocrine disorders. We report a 67-year-old woman with Cushing's syndrome due to left adrenal adenoma associated with myelolipoma. The patient underwent laparoscopic left adrenalectomy and pathological examination revealed an adrenocortical adenoma associated with myelolipoma. To the best of our knowledge, 25 cases of endocrine dysfunction associated with myelolipoma have been reported in the English and Japanese literature. We review and discuss the pathogenesis of adrenal myelolipoma.

Adenoma↗
PubMed2005

Presentation and therapy of myelolipoma.

BACKGROUND: Adrenal myelolipoma is a rare and benign, hormonally inactive tumor frequently discovered incidentally. Because of the increasing rate of detection of adrenal myelolipoma, use of the correct diagnostic examination and treatment, with respect to surgical excision or regular controls, is continually gaining importance. We report herein on the largest series of surgically treated patients with adrenal myelolipoma from a single institute. METHODS: The clinical charts of 12 patients who underwent adrenal surgery for myelolipoma were reviewed. Follow up was carried out by means of re-examination or by personal contact with the primary physician. It was possible to perform a re-examination on eight of the 12 patients 6.9 years after the operation: one patient had died 5.2 years after surgery. RESULTS: Diagnosis of adrenal myelolipoma was made incidentally in seven patients, and during examination for reasons of unspecific abdominal or flank pain in five patients. A transabdominal approach was taken in five patients, a subcostal approach was taken in three patients and a translumbal approach was taken in four patients. At the follow up, all formerly symptomatic patients were free of symptoms. No recurrence could be seen; however, in one patient a contralateral adrenal myelolipoma had developed. CONCLUSIONS: Symptomatic tumors, growing tumors or tumors larger than 10 cm should be excised surgically by means of an endoscopic or conventional approach, depending on the size. Surgical indication should not be liberalized by the introduction of more gentle operative techniques such as the translumbal or the endoscopic approach. A close follow up should be maintained in the case of patients free of symptoms, and for those with a definite diagnosis from imaging procedures.

Adrenal Gland Neoplasms↗
PubMed2006

Giant adrenal myelolipoma associated with small bowel leiomyosarcoma: a case report.

Adrenal myelolipoma is a rare, benign and biochemically inactive tumor. It is usually diagnosed incidentally by radiological methods and is known to be associated with obesity, hypertension, endocrinological disorders and some malignancies. We report herein the association of a myelolipoma with a gastrointestinal stromal tumor. To our knowledge this is the first report of such an association to date. A 67-year-old male patient admitted to our clinic with abdomimal pain and fever; he had a history of hypertension and diabetes mellitus. In physical examination, a mass involving the right quadrants was palpated. Computerized tomography revealed a right retroperitoneal mass, probably originating from the kidney or cecum. In laparotomy, the tumor (12 cm radius and 1500 g) localized on the superior of right kidney was excised. Abdomen exploration revealed another mass with 10 cm radius 100 cm distal to the ligamentum of Treitz and segmental jejunal resection and anastomosis were applied. The pathological diagnosis was reported as myelolipoma for the retroperitoneal mass and leiomyosarcoma for the jejunal mass. Myelolipoma is a benign tumor, involving mature fat and hematopoietic stem cells. Pathogenesis is still not clear and the microscopical characteristics are hematopoietic, lipoid, and reticuloid cells and megakaryocytes. Myelolipomas are reported to be associated with some other malignancies (especially renal), but this is the first report showing the association with a leiomyosarcoma. Therefore, leiomyosarcoma should also be one of the possible associations kept in mind by the physician in the diagnosis and treatment of myelolipomas.

Adrenal Gland Neoplasms↗
PubMed1995

Clinical spectrum of adrenal myelolipoma: analysis of 8 tumors in 7 patients.

Adrenal myelolipoma is a rare, benign, and biochemically nonfunctioning tumor. Most patients with adrenal myelolipoma are asymptomatic and the condition is discovered incidentally at autopsy. Review of the literature reveals 70 patients with surgically documented adrenal myelolipoma. We report 7 additional patients with 8 adrenal myelolipomas, of whom 5 underwent surgical removal because of tumor size, unusual imaging features or symptomatology. Of our patients 4 had symptomatic lesions requiring surgical removal, while 1 had an adrenal myelolipoma that was discovered intraoperatively. Of the remaining 2 patients 1 had a small (3 cm.) lesion discovered incidentally on excretory urography and 1 had bilateral adrenal myelolipoma discovered incidentally on computerized tomography.

Adrenal Gland Neoplasms↗
PubMed1994

Giant extra-adrenal myelolipoma.

Myelolipomas are rare, benign tumors composed in varying proportions of adipose tissue and hematopoietic cells. Myelolipomas are typically found in the adrenal gland, but uncommon cases of extra-adrenal sites have been reported, primarily presacrally. Extra-adrenal myelolipomas must be distinguished from masses of extramedullary hematopoiesis, which are also composed of hematopoietic elements but are associated with anemia and marked bone marrow hyperplasia. Also, if the distinction between extra-adrenal myelolipomas and malignant tumors such as liposarcoma were made before surgery, an invasive procedure might be avoided. We report the case of an extremely large, asymptomatic, perirenal myelolipoma in a 66-year-old diabetic woman. We also review the literature on extra-adrenal myelolipomas.

Aged↗
PubMed1987

Presacral myelolipoma.

Extramedullary tumors of hematopoietic tissue can be divided into two categories: those associated with a bone marrow disorder, myelolipomas which have no apparent relation to a bone marrow disorder. The etiology of myelolipoma is unknown and the vast majority are adrenal in location. Extramedullary myelolipomas are usually presacral in location and none of the approximately ten reported cases has been diagnosed before surgery. Although radiographic and ultrasound features of myelolipomas are relatively characteristic, they are not specific and biopsy is required for diagnosis. Microscopic diagnosis requires demonstration of benign lipomatous tissue admixed with cellular hematopoietic tissue resembling normal bone marrow. This study reports a patient with presacral myelolipoma and an unusual finding of urinary retention and azotemia associated with compression of urinary bladder neck by the myelolipoma.

Female↗
PubMed1989

Adrenal myelolipoma: a case report and review of the literature.

A case of a 73 year old man who was preoperatively diagnosed as possibly having myelolipoma is presented herein. Adrenal myelolipoma is an uncommon benign tumor of which only 74 foreign cases and 43 Japanese cases have been previously reported in the literature dealing with surgically removed myelolipomas. The most consistent complaint of the adrenal myelolipoma sufferer is abdominal pain, caused by hemorrhaging in the tumor, and many cases are associated with obesity, hypertension and/or diabetes mellitus. There is now an increasing number of such cases being diagnosed during abdominal scanning with ultrasonography or computerized tomography for unrelated problems, whereupon the myelolipoma is usually surgically resected. With the availability of modern scanning techniques and fine needle biopsy, however, it should be possible to adopt a more conservative approach to the management of asymptomatic adrenal myelolipoma. Nevertheless, symptomatic or large tumors, must be removed since there is a high risk of spontaneous hemorrhage.

Adrenal Gland Neoplasms↗
PubMed2000

Complete sequencing and mRNA expression analysis of the MEN-I gene in adrenal myelolipoma.

The molecular pathogenesis of adrenal myelolipoma is unclear. Endocrine activity of these tumors and association with other endocrine tumors have stimulated the hypothesis that it may belong to the group of sporadic tumors caused by defects of the gene responsible for multiple endocrine neoplasia type I (MEN-I). DNA of blood and tumoral sections from two patients with adrenal myelolipoma were analyzed by examination of variable number of tandem repeats (VNTR) loci PYGM, D11S987, D11S480, and D11S449 on chromosome 11q13 and by complete direct DNA sequencing of all coding exons and splice junctions of the MEN-I gene. Menin expression was examined by RT-PCR. RT-PCR did not detect menin expression in one adrenal myelolipoma. No loss of heterozygozity on chromosome 11q13 was identified. Intragenic heterozygozity was retained in codon 418 of the menin gene in both patients. No mutation was identified in the coding exons of the menin gene. Complete DNA sequencing yielded no hint that defects of the MEN-I gene are responsible for the formation of adrenal myelolipomas. Adrenal myelolipomas do not share the loss of heterozygozity on chromosome 11q13 observed in some benign adenomatous and many malignant adrenocortical tumors.

Adrenal Gland Neoplasms↗
PubMed2004

Adrenal myelolipoma associated with adenoma.

The association of an adrenal myelolipoma with a non-functioning adenoma is very rare. Herein, we report on such a case in an asymptomatic 64-year-old woman. To the best of our knowledge, there is only one other case of non-functioning adrenocortical adenoma associated with myelolipoma in the same gland. Furthermore, only two other adenomas (mineral corticoids and corticosteroid producing tumors) associated with myelolipomas have been reported, and both myelolipomas were less than 1 cm in size. In conclusion, the 8.5 cm myelolipoma in our case is the largest compared with the three previously reported ones.

Adenoma↗
PubMedDate not supplied

Spontaneous adrenal and hepatic myelolipomas in the common marmoset.

Myelolipomas occurring simultaneously in the adrenal and liver were found in a 2.7-yr-old, bred female common marmoset (Callithrix jacchus). The animal bore single adrenal and multiple hepatic myelolipomas. The adrenal myelolipoma consisted of mature adipose cells and focal collections of normal hematopoietic elements and was unencapsulated. In the liver, the myelolipomas, which were partially encapsulated, included a large amount of hematopoietic tissue and adipose cells that resembled normal bone marrow cells in various ways. Additionally, one of the multiple nodules contained several bony spicules with osteoblasts. Furthermore, there were invasive figures of hematopoietic cells, such as myeloblasts, erythroblasts, and megakaryocytes, in the hepatic sinusoids around the lesions. Thus, this case has some unusual morphological characteristics and is the first report, to our knowledge, on multiorganic myelolipomas in common marmosets.

Adrenal Gland Neoplasms↗