Primary Adrenal Leiomyosarcoma in an Aged Japanese Woman: A Rare Case Report

Aoki, Chie; Tanaka, Seiichi; Suzuki, Kunihiro; Sagara, Masaaki; Aso, Yoshimasa

doi:10.4172/2165-7920.1000935

Cited by 3 publications

(13 citation statements)

References 13 publications

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“…Regarding hormonal secretion from the tumor, one case presented with markedly high levels of serum neuron specific enolase (NSE) which were normalised after excision (41). One case had elevated levels of NSE, IL-2 receptor, thymidine kinase activity and adrenocorticotropic hormone (ACTH) (36). One patient showed elevated serum CA125 values during workup (67).…”

Section: Symptoms Diagnosismentioning

confidence: 99%

“…Seven cases were depicted as avid masses in PET-CT (34,38,41,51,53,54,62), 5 cases were described as negative in MIBG scintigraphy (41)(42)(43)(44)64) and 2 in aldosterone scintigraphy (41,42). One case was tested through 67 gallium scintigraphy and was found positive (36).…”

Section: Risk Factorsmentioning

confidence: 99%

“…From these cases, 5 patients proceeded with surgical excision, one of them was deemed inoperable during exploratory laparotomy (59), and the remaining 4 were not qualified as surgical candidates due to either local extension or metastatic disease (32,34,38,51). Regarding diagnosis through biopsy specimen, 5 specimens revealed the presence of a spindle cell neoplasm, with (57) or without smooth muscle differentiation (43,55,47,49) and in 4 cases the diagnosis of a malignant spindle cell neoplasm was rendered (30,32,36,67).…”

Section: Risk Factorsmentioning

confidence: 99%

“…One case that was deemed inoperable underwent thoracic surgery for metastases (38) and three cases underwent autopsy examination (3, 59). One patient received preoperative chemotherapy (65), 5 patients received combination chemotherapy with radiotherapy postoperatively (39,44,45,50,73), 5 patients received adjuvant chemotherapy (36,49,54,61,71), 2 patients received radiotherapy postoperatively (35,64) and one patient received combination chemotherapy RT and radiofrequency ablation (RFA) postoperatively (42). One patient received combination chemotherapy and RT only in vivo 34: 2233-2248 (2020) (51), 2 cases underwent chemotherapy only (32,38) and one patient received RT only (34).…”

Section: Risk Factorsmentioning

confidence: 99%

“…One patient was treated with both regimens sequentially (73). One patient was treated with doxorubicin-based chemotherapy and then with cisplatin (42) and one other with receptor tyrosine kinase (RTK) and mechanistic target of rapamycin (mTOR) inhibitors (36). The patient treated with neoadjuvant chemotherapy received paclitaxel (65).…”

Section: Risk Factorsmentioning

confidence: 99%

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Review of the Literature on Leiomyoma and Leiomyosarcoma of the Adrenal Gland: A Systematic Analysis of Case Reports

Σακελλαρίου

Dellaportas

Peppa

et al. 2020

In Vivo

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Background/Aim: To date few cases of smooth muscle-derived tumors of the adrenal gland have been reported and their treatment remains a medical challenge. The aim of this manuscript was to systematically review the literature and present the tumor characteristics and their management in order to provide a standardized approach to their diagnosis and management. Materials and Methods: We searched five databases (PubMed, Scopus, Elsevier, ResearchGate, Google scholar) for relevant articles published until March 2020. Results: Twenty-two cases of adrenal leiomyoma, four cases of adrenal smooth muscle tumor and forty-five cases of adrenal leiomyosarcoma were included. Conclusion: We present the demographic, clinical, radiological, pathological and oncological characteristics and prognosis of tumors of the adrenal gland arising from smooth muscle cells, as well as describe the common clinical investigations and therapeutic modalities that have been reported as part of their management.Adrenal tumors can be detected either incidentally or after investigation of signs and symptoms that they evoke. Generally, they can be histologically categorized as primary tumors of the adrenal cortex or medulla, metastases from other primary malignancies and other tumors of the adrenal gland which originate from connective tissue cells. Adrenal cortical tumors are comprised by (asymmetric) adrenal cortical hyperplasia, adenoma, myelolipoma, adrenocortical carcinoma, oncocytic adrenocortical neoplasms; medullary tumors are comprised by pheochromocytoma, ganglioneuroma, neuroblastoma, ganglioneuroblastoma and teratoma. Connective tissue origin tumors are rarer and are comprised mainly by haemangioma and lymphangioma with a few cases of leiomyoma, lipoma, hemangiosarcoma, lymphoma, leiomyosarcoma and liposarcoma, as reported in the literature. Patients with primary malignancies in lung, breast, kidney, colon, oesophagus, pancreas, liver, stomach or with melanoma can present with adrenal metastases spread from these tumors, which can commonly be found bilaterally (1).Leiomyomas (LM) and leiomyosarcomas (LMS) are mesenchymal tumors that derive from smooth muscle cells. Their benign -leiomyoma or malignant -leiomyosarcoma characterization depends on nuclear atypia and/or pleomorphism, mitotic activity and tumor cell necrosis (2). Leiomyomas and leiomyosarcomas of the adrenal gland comprise a very small percentage of the gland's pathology with very little data available in the literature. The aim of this manuscript was to systematically review the literature and present demographic, clinical, radiological, pathological and oncological characteristics and prognosis of tumors of the adrenal gland arising from smooth muscle cells, as well as describe the common clinical investigations and therapeutic modalities that have been reported as part of their management.

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Section: Symptoms Diagnosismentioning

confidence: 99%

Section: Risk Factorsmentioning

confidence: 99%

Section: Risk Factorsmentioning

confidence: 99%

Section: Risk Factorsmentioning

confidence: 99%

Section: Risk Factorsmentioning

confidence: 99%

See 3 more Smart Citations

Review of the Literature on Leiomyoma and Leiomyosarcoma of the Adrenal Gland: A Systematic Analysis of Case Reports

Σακελλαρίου

Dellaportas

Peppa

et al. 2020

In Vivo

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Primary adrenal leiomyosarcoma with inferior vena cava extension in a 70-year-old man

et al. 2019

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Primary adrenal leiomyosarcoma, a malignant soft tissue tumour originating from the smooth muscle of the inferior vena cava and adrenal vein, is rarely described in the literature. Cases are often diagnosed at an advanced stage as the tumour is not hormone-producing. We describe a 70-year-old man who presented with lower extremity swelling and abdominal varices and was subsequently found to have a large adrenal mass on imaging. Our case is among the few reported primary adrenal leiomyosarcomas in which a CT-guided biopsy was used to aid in diagnosis.

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Outcomes and Follow-Up Trends in Adrenal Leiomyosarcoma: A Comprehensive Literature Review and Case Report

Mongardini,

Paolicelli,

Catauro

et al. 2024

JCM

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Background: Leiomyosarcoma (LMS) originating from the adrenal gland is exceedingly rare, constituting a minute fraction of soft tissue sarcomas. Due to its rarity, with less than 50 documented cases in English medical literature, the diagnosis and management of adrenal LMS remain challenging. The aim of this study was to perform a review of the literature, in order to evaluate the prognosis of these rare cancers and report our specific case. Methods: A systematic review of the literature was conducted using PubMed, Web of Science, Google Scholar, and Scopus databases, up to December 2020. The search utilized MeSH terms such as “Adrenal Gland Neoplasms,” “Leiomyosarcoma,” “Adrenalectomy,” and “Smooth Muscle Tumor.” The inclusion criteria focused on studies reporting patients with a histopathological diagnosis of adrenal leiomyosarcoma. The PRISMA guidelines were followed to ensure a comprehensive analysis. Results: Out of 63 identified studies, 43 met the inclusion criteria and were reviewed. These studies highlighted the rarity and aggressive behavior of adrenal leiomyosarcoma. Surgical excision remains the cornerstone of treatment, often complemented by adjuvant therapies. The reviewed case involved a 52-year-old woman who underwent a right laparoscopic adrenalectomy for a 9 × 7 × 6 cm grade 3 leiomyosarcoma. Despite subsequent adjuvant chemotherapy, hepatic metastases were detected, illustrating the aggressive nature of the disease. The literature underscores the importance of histopathological analysis and long-term surveillance for managing disease progression. Conclusions: Optimal management of adrenal leiomyosarcoma requires a multidisciplinary approach and meticulous follow-up. The rarity of the disease poses challenges for standardizing treatment, but surgical excision and tailored adjuvant therapies show promise. Further research is essential to refine treatment strategies and improve prognosis for this rare malignancy.

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Primary Adrenal Leiomyosarcoma in an Aged Japanese Woman: A Rare Case Report

Cited by 3 publications

References 13 publications

Review of the Literature on Leiomyoma and Leiomyosarcoma of the Adrenal Gland: A Systematic Analysis of Case Reports

Review of the Literature on Leiomyoma and Leiomyosarcoma of the Adrenal Gland: A Systematic Analysis of Case Reports

Primary adrenal leiomyosarcoma with inferior vena cava extension in a 70-year-old man

Outcomes and Follow-Up Trends in Adrenal Leiomyosarcoma: A Comprehensive Literature Review and Case Report

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