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Pradhan S, Voni SK, Sadangi S, Kalita J, Panda AK, Agarwal SK, Mohapatra D, Panda SK, Dash SS. Bilateral Extra-adrenal Myelolipoma: A Rare Case with Literature Review. Indian J Surg Oncol 2025; 16:427-431. [PMID: 40337019 PMCID: PMC12052662 DOI: 10.1007/s13193-024-02121-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/19/2024] [Accepted: 10/17/2024] [Indexed: 05/09/2025] Open
Abstract
Myelolipoma is a benign tumour comprising both hematopoietic tissue and normal adipose tissue. It is a rare tumour found incidentally in adrenal glands on imaging, rarely reported in the extra-adrenal/perirenal region with less than 50 cases being reported in the literature. Even rarely, it presents bilaterally. We report a case of a 65-year-old male with bilateral extra-adrenal myelolipoma who underwent en bloc excision of bilateral tumours diagnosed preoperatively by radiological imaging. We reviewed the literature to look into the incidence, aetiology, imaging characteristics, diagnosis, treatment and follow-up in patients with extra-adrenal myelolipomas.
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Affiliation(s)
- Snehasis Pradhan
- Department of Surgical Oncology, IMS & SUM Hospital, Siksha ‘O’ Anusandhan, 751003 Bhubaneswar, India
| | - Sujan Kumar Voni
- Department of Surgical Oncology, IMS & SUM Hospital, Siksha ‘O’ Anusandhan, 751003 Bhubaneswar, India
| | - Sudam Sadangi
- Department of Surgical Oncology, IMS & SUM Hospital, Siksha ‘O’ Anusandhan, 751003 Bhubaneswar, India
| | - Jubraj Kalita
- Department of Surgical Oncology, IMS & SUM Hospital, Siksha ‘O’ Anusandhan, 751003 Bhubaneswar, India
| | - Adya Kinkar Panda
- Department of Radiodiagnosis, IMS & SUM Hospital, Siksha ‘O’ Anusandhan, 751003 Bhubaneswar, India
| | - Sunil Kumar Agarwal
- Department of Surgical Oncology, IMS & SUM Hospital, Siksha ‘O’ Anusandhan, 751003 Bhubaneswar, India
| | - Debahuti Mohapatra
- Department of Pathology, IMS & SUM Hospital, Siksha ‘O’ Anusandhan, 751003 Bhubaneswar, India
| | - Sangram Keshari Panda
- Department of Surgical Oncology, IMS & SUM Hospital, Siksha ‘O’ Anusandhan, 751003 Bhubaneswar, India
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Shimoda H, Kijima T, Takada-Owada A, Ishida K, Kamai T. A case of perirenal extra-adrenal myelolipoma mimicking liposarcoma. Urol Case Rep 2023; 50:102523. [PMID: 37664534 PMCID: PMC10468332 DOI: 10.1016/j.eucr.2023.102523] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/26/2023] [Accepted: 08/13/2023] [Indexed: 09/05/2023] Open
Abstract
Myelolipoma is a benign tumor composed of mature adipose tissue and normal hematopoietic components. It usually occurs in the adrenal glands but rarely in the extra-adrenal region. However, it is difficult to differentiate extra-adrenal myelolipoma from well-differentiated liposarcoma on the basis of the radiological findings. We report the case of a 66-year-old male with perirenal and extra-adrenal myelolipoma who underwent radical tumor resection with nephrectomy after a preoperative diagnosis of liposarcoma. Intraoperative assessment by the surgeon and intraoperative pathological evaluation are important considering the divergent prognoses of myelolipoma and liposarcoma.
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Affiliation(s)
- Hiroki Shimoda
- Department of Urology, Dokkyo Medical University, 880 Kitakobayashi, Mibu, Shimotsuga, Tochigi, 321-0293, Japan
| | - Toshiki Kijima
- Department of Urology, Dokkyo Medical University, 880 Kitakobayashi, Mibu, Shimotsuga, Tochigi, 321-0293, Japan
| | - Atsuko Takada-Owada
- Department of Diagnostic Pathology, Dokkyo Medical University, 880 Kitakobayashi, Mibu, Shimotsuga, Tochigi, 321-0293, Japan
| | - Kazuyuki Ishida
- Department of Diagnostic Pathology, Dokkyo Medical University, 880 Kitakobayashi, Mibu, Shimotsuga, Tochigi, 321-0293, Japan
| | - Takao Kamai
- Department of Urology, Dokkyo Medical University, 880 Kitakobayashi, Mibu, Shimotsuga, Tochigi, 321-0293, Japan
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Zhanghuang C, Long N, Yang Z, Xie Y. Bilateral adrenal giant medullary lipoma combined with disorders of sex development: a rare case report and literature review. Front Oncol 2023; 13:1210679. [PMID: 37681022 PMCID: PMC10482419 DOI: 10.3389/fonc.2023.1210679] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/23/2023] [Accepted: 07/31/2023] [Indexed: 09/09/2023] Open
Abstract
Bilateral adrenal myelolipoma is rare in clinics and patients with disorders of sex development (DSDs). One case was reported in our center. A 45-year-old patient was admitted to the hospital after discovering a left abdominal mass for more than a year and worsening abdominal pain for 18 days. An imaging examination showed bilateral adrenal masses. Physical examination showed clitoris hypertrophy with patelliform changes, thick and dense pubic hair, normal development of bilateral labia majora without labia minora, and urethral opening. After the relevant preoperative examinations, bilateral adrenal mass resection was performed under general anesthesia. The postoperative pathology confirmed adrenal myelolipoma. The incision healed well without recurrence over 10 years after the operation. Her enlarged clitoris decreased in size. This case report has a detailed diagnosis and treatment process and sufficient examination results. It can provide a reference for diagnosing and treating patients with bilateral adrenal myelolipoma and DSD and reduce the risk of misdiagnosis and mistreatment.
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Affiliation(s)
- Chenghao Zhanghuang
- Department of Urology, Kunming Children’s Hospital, Yunnan Province Clinical Research Center for Children’s Health and Disease, Kunming, China
- Yunnan Key Laboratory of Children’s Major Disease Research, Yunnan Clinical Medical Center for Pediatric Diseases, Kunming Children’s Hospital, Kunming, China
| | - Na Long
- Special Ward, Kunming Children’s Hospital, Yunnan Province Clinical Research Center for Children’s Health and Disease, Kunming, China
| | - Zhen Yang
- Department of Oncology, Yunnan Children Solid Tumor Treatment Center, Kunming Children’s Hospital, Kunming, China
| | - Yucheng Xie
- Department of Pathology, Kunming Children’s Hospital, Kunming, China
- Department of Pathology, The Second People’s Hospital of Yunnan Province, Kunming, China
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Xu C, Kasajima A, Novotny A, Friess H. Meta-analysis of reported presacral myelolipomas, including a report of a new case. J Med Case Rep 2023; 17:31. [PMID: 36721209 PMCID: PMC9890845 DOI: 10.1186/s13256-022-03746-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/17/2022] [Accepted: 12/27/2022] [Indexed: 02/02/2023] Open
Abstract
BACKGROUND Presacral myelolipomas form a rare disease and are often found incidentally in imaging diagnostics. CASE PRESENTATION In this study, we report the case of a 71-year-old caucasian female with an incidental finding of a retroperitoneal tumor on magnetic resonance imaging scan. This report aimed at presenting the clinical course of this patient with emphasis on analysis of pathological, clinical, and epidemiological features in a meta-analysis of reported cases. CONCLUSION Presacral myelolipomas are rare and its etiology remains unclear. Surgical resection is indicated in symptomatic lesions and lesions > 4 cm. More clinical and pathological research on this rare entity is warranted.
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Affiliation(s)
- Congde Xu
- grid.6936.a0000000123222966Klinikum rechts der Isar, Klinik und Poliklinik für Chirurgie, TU München, Ismaninger Straße 22, 81675 Munich, Deutschland
| | - Atsuko Kasajima
- grid.6936.a0000000123222966Klinikum rechts der Isar, Institut für Allgemeine Pathologie und Pathologische Anatomie, TU München, Ismaninger Straße 22, 81675 Munich, Deutschland
| | - Alexander Novotny
- grid.6936.a0000000123222966Klinikum rechts der Isar, Klinik und Poliklinik für Chirurgie, TU München, Ismaninger Straße 22, 81675 Munich, Deutschland
| | - Helmut Friess
- grid.6936.a0000000123222966Klinikum rechts der Isar, Klinik und Poliklinik für Chirurgie, TU München, Ismaninger Straße 22, 81675 Munich, Deutschland
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Cao J, Huang X, Cui N, Wang X, You C, Ni X, Gao X, Wang J, Liu T. Surgical Management and Outcome of Extra-adrenal Myelolipomas at Unusual Locations: A Report of 11 Cases in a Single Center. J Bone Oncol 2022; 35:100438. [PMID: 35721369 PMCID: PMC9198452 DOI: 10.1016/j.jbo.2022.100438] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/19/2022] [Revised: 06/04/2022] [Accepted: 06/04/2022] [Indexed: 11/28/2022] Open
Abstract
The first case series with eleven consecutive patients diagnosed with EAMs in the spinal or intraosseous regions. EAMs involving the thoracic vertebral body or the humerus have not previously been reported in the English literature. The surgical outcome and prognosis of EAMs are excellent and surgery can serve as the method of radical treatment. Purpose Extra-adrenal myelolipomas (EAMs) are rare benign tumors composed of both mature adipose and hematopoietic tissues with unclear etiology. There have been only sporadic case reports about the clinical characteristics and management of EAMs. Here we present our experience and practice in the clinical diagnosis and treatment of 11 consecutive patients with EAMs. Method We retrospectively reviewed 11 consecutive patients, who received surgeries in our department and were confirmed as having EAMs by postoperative histopathology from April 2016 to December 2021. Clinical information and follow-up data of all patients were collected and analyzed afterwards. Results Of the 11 EAM patients (7 male and 4 female) with a mean age of 47.6 years, 3 were asymptomatic and 8 were symptomatic with a mean symptom duration of 6.07 months. EAMs were found in the thoracic spine in 4 cases, paravertebral mediastinal regions in 3 cases, ilium in 2 cases, humerus in 1 case, and rib in 1 case. All patients were initially misdiagnosed as other tumors by radiologists. All 11 patients received gross total excision or curettage with a mean intraoperative blood loss of 781.82 ± 1143.3 ml and a mean operation duration of 180.91 ± 98.41 min. Patients’ Frankel scores and Karnofsky Performance Status score were improved or at least preserved postoperatively. No significant complications occurred postoperatively. All the 11 patients survived, and no local recurrence or distant metastasis occurred during the mean follow-up period of 42.0 months. Conclusion The surgical outcome and prognosis of EAMs are excellent and surgery can serve as the method of radical treatment.
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Affiliation(s)
- Jiashi Cao
- Department of Orthopedics, No. 455 Hospital of the Chinese People's Liberation Army, The Navy Medical University, No. 338 Huaihai West Road, Shanghai 200052, China
- Department of Orthopaedic Oncology, Changzheng Hospital of the Navy Medical University, No. 415 Fengyang Road, Shanghai 200003, China
| | - Xing Huang
- Department of Orthopaedic Oncology, Changzheng Hospital of the Navy Medical University, No. 415 Fengyang Road, Shanghai 200003, China
| | - Na Cui
- Department of Pathology, Changzheng Hospital of the Navy Medical University, No. 415 Fengyang Road, Shanghai 200003, China
| | - Xiang Wang
- Department of Radiology, Changzheng Hospital of the Navy Medical University, No. 415 Fengyang Road, Shanghai 200003, China
| | - Chaoqun You
- Department of Orthopaedic Oncology, Changzheng Hospital of the Navy Medical University, No. 415 Fengyang Road, Shanghai 200003, China
- Clinical Medical College of Weifang Medical University, No.7166 Baotong West Street, Weifang, Shandong 261000, China
| | - Xiangzhi Ni
- Department of Orthopaedic Oncology, Changzheng Hospital of the Navy Medical University, No. 415 Fengyang Road, Shanghai 200003, China
| | - Xin Gao
- Department of Orthopaedic Oncology, Changzheng Hospital of the Navy Medical University, No. 415 Fengyang Road, Shanghai 200003, China
- Corresponding authors.
| | - Jing Wang
- Department of Orthopaedic Oncology, Changzheng Hospital of the Navy Medical University, No. 415 Fengyang Road, Shanghai 200003, China
- Corresponding authors.
| | - Tielong Liu
- Department of Orthopaedic Oncology, Changzheng Hospital of the Navy Medical University, No. 415 Fengyang Road, Shanghai 200003, China
- Corresponding authors.
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