Congenital Adrenal Hyperplasia with Combined 21-hydroxylase deficiency and 17α-hydroxylase/17,20-lyase deficiency: An undervirilized male.

21-hydroxylase deficiency stands as the most prevalent form of congenital adrenal hyperplasia, primarily resulting from mutations in the CYP21A2 gene. On the other hand, mutations within the CYP17A1 gene lead to 17α-hydroxylase/17,20-lyase enzyme deficiencies. The scarcity of 17-OH deficiency is noteworthy, accounting for less than 1% of all congenital adrenal hyperplasia cases. The male patient, born from a first-degree cousin marriage, exhibited several symptoms, including left undescended testis, micropenis, penile chord, left sensorineural hearing loss, and gynecomastia. He reported micropenis as a concern at the age of 13.5 years. His hormone profile revealed high levels of serum 17-hydroxyprogesterone, progesterone, and pregnenolone. In this case with a 46 XY karyotype, suspicions arose regarding Cytochrome P450 oxidoreductase deficiency due to ambiguous genitalia and an atypical hormone profile. Analysis unveiled two distinct homozygous and pathogenic variants in the CYP21A2 and CYP17A1 genes. Notably, mineralocorticoid precursors escalated, while cortisol and sex steroid precursors decreased during the high (250 mcg) dose ACTH stimulation test. The mutation c.1169C > G (p.Thr390Arg) in CYP17A1, which is the second documented case in literature, stands out due to its unique set of accompanying features. Mutations occurring in CYP21A2 and CYP17A1 result in complete or partial enzyme deficiencies, and the detection of homozygous mutations in two different enzyme systems within the steroidogenic pathway is noteworthy.

The Scar-Hidden Surgery on Gynecomastia: Experiences from a Single-Institutional Large Case Series.

BACKGROUND: To summarize the experiences on the mastoscopic subcutaneous mastectomy for gynecomastia by "nine-step method" based on the "5S" goal and standardize this operation. PATIENTS AND METHODS: Between January 1, 2002, and October 31, 2021, a total of 2035 breasts of 1082 male patients with gynecomastia, of which 129 patients with one side, were underwent mastoscopic subcutaneous mastectomy. The follow-up endpoint was 3 months after surgery. RESULTS: All patients were successfully completed the operation, and none of them was transferred to open operation. The operation time for unilateral breast was 12-28 min, and the average time was 17.7 ± 6.2 min. The amount of bleeding during unilateral operation was very small, about 5-10 ml. The total drainage volume was 5-50 ml after the operation, and the drainage tube was removed in 3-5 days. The epidermal necrosis occurred in 0.3% nipple. 0.2% chest wall had a little ecchymosis in the supero-medial region of the breast. All patients had the normal feeling of nipples and areola, the smoothing and symmetrical chest wall, and the natural contour. There was no recurrence during the follow-up period. CONCLUSIONS: The mastoscopic subcutaneous mastectomy for gynecomastia by "nine-step method" based on the "5S" goal has a short operation time, few surgical complications and good esthetics. It achieves the "5S" goals on the complete removal of glandular tissue (sweeping), small and scar-hidden incision are small (scarless), good symmetry of bilateral chest wall (symmetry), normal chest shape (shape), and smoothing chest wall (smoothing). LEVEL OF EVIDENCE III: The journal asks authors to assign a level of evidence to each article. For a complete description of Evidence-Based Medicine ratings, see the Table of Contents or the online Instructions for Authors at www.springer.com/00266 .

Deciphering the molecular landscape: evolutionary progression from gynecomastia to aggressive male breast cancer.

BACKGROUND: Gynecomastia denotes the benign proliferation of glandular breast tissue and stands as a recognized risk factor for male breast cancer. Nonetheless, the underlying carcinogenic mechanisms orchestrating the progression from gynecomastia to cancer remain poorly understood. METHODS: This study employed single-cell RNA sequencing (scRNA-seq) to meticulously dissect the cellular landscape of gynecomastia and unravel potential associations with male breast cancer at a single-cell resolution. Pseudotime and evolutionary analyses were executed to delineate the distinct features characterizing gynecomastia and male breast cancer. The TCGA database, along with cell-cell communication analysis and immunohistochemistry staining, was harnessed to validate differential gene expression, specifically focusing on CD13. RESULT: From the copy number variation profiles and evolutionary tree, we inferred shared mutation characteristics (18p+ and 18q+) underpinning both conditions. The developmental trajectory unveiled an intriguing overlap between gynecomastia and malignant epithelial cells. Moreover, the differential gene CD13 emerged as a common denominator in both gynecomastia and male breast cancer when compared with normal mammary tissue. Cell-cell interaction analysis and communication dynamics within the tumor microenvironment spotlighted distinctions between CD13+ and CD13- subsets, with the former exhibiting elevated expression of FGFR1-FGF7. CONCLUSIONS: Our investigation provides novel insights into the evolutionary progression from gynecomastia to male breast cancer, shedding light on the pivotal role of CD13 in driving this transition. The identification of CD13 as a potential therapeutic target suggests the feasibility of CD13-targeted interventions, specifically tailored for male breast cancer treatment.

Looking Beyond the Usual Suspects: A Rare Case of Teriparatide-Induced Gynecomastia.

Teriparatide, an osteoanabolic agent, is a biosynthetic analogue of the 1-34 amino acids of human parathyroid hormone (PTH) used for the treatment of osteoporosis. It is typically well-tolerated; common side effects include headaches, arthralgias, nausea, and dizziness. In this report, we present a case of gynecomastia occurring shortly after initiating teriparatide therapy, associated with nipple sensitivity and breast tenderness. Secondary workup for various causes of gynecomastia was unremarkable. Finally, a decision was made to discontinue teriparatide due to the patient's concerns. The nipple sensitivity started improving shortly afterward, with complete resolution of gynecomastia 4 months later. Although this unusual side effect has been reported as a possibility in postmarketing studies, a chronological report on the occurrence of teriparatide-induced gynecomastia and its complete resolution after discontinuing teriparatide has not yet been published in the literature.

Gynecomastia in adolescent males: current understanding of its etiology, pathophysiology, diagnosis, and treatment.

Gynecomastia (GM) is a common and continuously evolving condition that commonly occurs during adolescence. It is the source of significant embarrassment and psychological stress in adolescent males. GM is characterized by enlargement of the male breast due to the proliferation of glandular ducts and stromal components. The main cause of GM during adolescence is physiological or pubertal GM, which is primarily attributed to an imbalance between estrogen and androgen activity. Physiological GM is typically transient and resolves within several months, although it may take several years to resolve. GM may also be caused by other pathological conditions and could be indicative of an endocrine disease. It is crucial to understand the pathogenesis of GM to distinguish it from normal developmental variants due to pathological causes. The aim of this review is to highlight the significance of GM during adolescence in terms of potential etiologies, clinical and laboratory diagnoses, and current management.

A Three-Dimensional Approach To Male Chest Enhancement: A Surgical Algorithm Based On 300 Cases.

Gynecomastia is defined as a benign condition of the male caused by tissue overgrowth (Blau and Hazani in Plast Reconstruct Surg 135(2): 425-432, 2015). Its incidence ranges widely in the world population, ranging from 32 to 65% (Innocenti et al. in Ann Plast Surg 78(5):492-496, 2017). Pseudogynecomastia is a condition characterized by deposits of adipose tissue with alteration of the profile of the male thorax. It appears clinically similar to gynecomastia (Hoyos et al. in Plast Reconstr Surg 147:1072-1083, 2021). Several classification systems that characterize the severity of male breast hypertrophy have been described in the literature, and many surgical algorithms have been formulated for its treatment (Holzmer et al. in Plast Reconstruct Surg-Global Open 8:e3161, 2020). The purpose of this original article is to provide a comprehensive surgical algorithm for the management of male chest enhancement based on severity, as defined by the Moschella scale (Tambasco et al. in J Plast Reconstruct Aesthet Surg 90:99-100, 2024). A total of 300 patients treated for bilateral breast hypertrophy are included and reviewed in this retrospective study. Patients have been diversified according to the Moschella scale. For each grade up to grade III, two subgroups were distinguished: A) pinch test less than 0.7 cm and B) pinch test greater than 0.7 cm. For Grade IV, we distinguished: subgroup A) where the distance between the inframammary fold and the nipple was < 3 cm; and subgroup B) where the distance between the inframammary fold and the nipple was > 3 cm. We developed an algorithm, based on this experience, to help to choose the best surgical techniques to perform a three-dimensional result. All patients were treated using multiple surgical techniques. In all cases, we made a reduction in the hypertrophy of the chest, obtaining the three dimensionality. Associate techniques include ultrasound-assisted liposuction (UAL) and helium plasma radiofrequency technology (HPRF). A round block mastectomy (RBm) or skin-reducing mastectomy T inverted (SRM Tinv) is reserved only in limited cases.Level of Evidence IV: This journal requires that authors assign a level of evidence to each article. For a full description of these Evidence-Based Medicine ratings, please refer to the Table of Contents or the online Instructions to Authors www.springer.com/00266 .

Bilateral Metastatic Gynecomastia from Small-Cell Lung Tumors in a Man: A Report of a Rare Case.

A case of a man with the recent onset of painful bilateral firm gynecomastia is reported. Mammography confirmed increased breast density. Biopsy characterized both masses as metastases of a small-cell lung tumor. This case highlights the atypical presentation and complements the literature regarding the rarity of breast metastases from small-cell lung cancer in men. Teaching point: Bilateral gynecomastia in a man with a long history of cigarette smoking should be considered with caution.

Persistent Gynecomastia due to Short-term Low-dose Finasteride for Androgenetic Alopecia.

We report a case of persistent gynecomastia in a healthy 20-year-old man after 1 month of low-dose finasteride. Finasteride was discontinued after 2 months, and gynecomastia was unchanged 5 months after drug withdrawal. The gynecomastia regressed but did not resolve after 6 months of treatment with raloxifene, a selective estrogen receptor modulator. One year later, bilateral mammoplasty was performed to remove the remaining breast tissue. Finasteride, a 5-alpha-reductase inhibitor, is widely used for the treatment of androgenetic alopecia. Gynecomastia is an expected side effect of this therapy given its mechanism of action. However, only 8 cases of gynecomastia have been reported with low-dose (1 mg daily) finasteride treatment since its approval for androgenetic alopecia in 1997. This raises the concern that gynecomastia resulting from low-dose finasteride is significantly underreported, causing inadequately informed patients. Further, because of the risk of gynecomastia, it is important for prescribing physicians to counsel patients regarding this complication and to consider early intervention when finasteride-induced gynecomastia first arises to prevent fibrosis and thus irreversible gynecomastia.

Gynecomastia Surgery in 4996 Male Patients Over 14 Years: A Retrospective Analysis of Surgical Trends, Predictive Risk Factors, and Short-Term Outcomes.

BACKGROUND: The high prevalence of benign male breast tissue enlargement (gynecomastia) has resulted in a marked increase of gynecomastia cases. While about one third of male adults experience some form of gynecomastia, gynecomastia surgery (GS) outcome research is limited to small study populations and single-center/-surgeon databases. In this study, we aimed to access the American College of Surgeons National Surgical Quality Improvement Program (ACS-NSQIP) database to identify preoperative risk factors for complications and investigate postoperative outcomes of GS. METHODS: In this retrospective study, we queried the ACS-NSQIP database from 2008 to 2021 to identify male adult patients who underwent GS. Postoperative outcomes involved the occurrence of any, surgical and medical complications, as well as reoperation, readmission, and mortality within a 30-day postoperative time period. Univariable and multivariable assessment were performed to identify risk factors for complications while adjusting for possible confounders. RESULTS: The study included 4,996 GS patients with a mean age of 33.7 ± 15 years and BMI of 28.2 ± 5.1 kg/m2. White patients constituted 54% (n = 2713) of the cohort, and 27% (n = 1346) were obese. Except for 2020, there was a steady increase in GS cases over the study period. Outpatient surgeries were most common at 95% (n = 4730), while general surgeons performed the majority of GS (n = 3580; 72%). Postoperatively, 91% (n = 4538) of patients were discharged home; 4.4% (n = 222) experienced any complications. Multivariable analysis identified inpatient setting (p < 0.001), BMI (p = 0.023), prior sepsis (p = 0.018), and bleeding disorders (p = 0.047) as independent risk factors for complications. CONCLUSION: In this study, we analyzed 4996 male adult GS patients from the ACS-NSQIP database, revealing an increased caseload and significant general surgeon involvement. Risk factors like bleeding disorders, inpatient status, and prior sepsis were linked to postoperative complications, while BMI was crucial for predicting adverse events. Overall, our findings may aid in enhancing patient care through advanced preoperative screening and closer perioperative management. LEVEL OF EVIDENCE III: This journal requires that authors assign a level of evidence to each article. For a full description of these Evidence-Based Medicine ratings, please refer to the Table of Contents or the online Instructions to Authors www.springer.com/00266 .

Pubertal gynecomastia incidence among 530,000 boys: a cross sectional population based study.

Background: Adolescent gynecomastia, a benign proliferation of male breast tissue, can lead to psychological issues during adolescence. The prevalence varies widely (4%-69%). The incidence peaks are during neonatal, pubertal, and senescent periods. Its affect on emotional well-being necessitates understanding and occasional intervention. This study aimed to determine the incidence of gynecomastia among male adolescents aged 12-15 years. Methods: A retrospective cross-sectional study utilized the Clalit Health Care Services database (2008-2021) with a population of approximately 4.5 million. Participants aged 12-15 years were included if diagnosed with gynecomastia (International classification of diseases-9 code 611.1) and having a body mass index (BMI) measurement and no obesity diagnosis (ICD9 code 278.0). Data analysis included incidence rates and associations with ethnicity, age, BMI, and socioeconomic status. Results: 531,686 participants included with an incidence of 1.08%. Of all participants, 478,140 had a BMI ≤ 25 with an incidence of 0.7%, and 0.25%-0.35% yearly, and 70% of gynecomastia patients were aged 13-14 years. The prevalence of gynecomastia differed between Jews (1.28%) and Arabs (0.67%), but the disparity diminished when socioeconomic status was considered. Conclusions: This unprecedented Population study establishes a definitive rate of true pubertal gynecomastia, revealing a lower yearly incidence as compared to previous reports. The higher observed prevalence among Jewish adolescents, may be caused due to complex interactions between different influencing factors. Understanding these dynamics can aid in formulating more targeted interventions and policy strategies to address gynecomastia's affect on adolescent well-being.

Infra-areolar subcutaneous mastectomy in the surgical treatment of adolescent gynecomastia.

BACKGROUND: Despite being a common condition in puberty, only 5-10% of pubertal gynecomastia need surgical treatment. Here the authors present their experiences with infra-areolar subcutaneous mastectomy in the surgical treatment of adolescent gynecomastia. METHODS: The records of patients who underwent infra-areolar subcutaneous mastectomy for adolescent gynecomastia between January 2004 and December 2021 were reviewed retrospectively. The patients' demographic data, complaints and clinical presentation, physical examination, laboratory and radiological findings, surgical management, and postoperative follow-ups were evaluated. The patients were evaluated according to the localization of the gynecomastia (unilateral/bilateral) and according to the patients' body mass index (BMI) (normal/overweight). RESULTS: A total of 21 boys with a mean age of 15 ± 1.4 years were operated by the senior author and infra-areolar subcutaneous mastectomy by a semilunar incision was performed for adolescent gynecomastia. The mean duration of the complaint was 24.2 ± 10.9 months. Gynecomastia was bilateral in 15 (71.5%) and unilateral in 6 (28.5%) of the patients. Sixteen patients (76.2%) were normal weight, 5 (23.8%) were overweight. Chromosomal anomaly and hypogonadism were detected in two patients. Pseudoangiomatous stromal hyperplasia was detected in the pathology of one patient. The mean follow-up time was 2.7 ± 1 years, seroma developed in 2 patients at the early postoperative period. Their long-term follow-up was uneventful without complication. CONCLUSIONS: The results of infra-areolar subcutaneous mastectomy are very promising in pubertal gynecomastia due to good skin elasticity and without obesity in the adolescent age group. We believe that this method provides an almost invisible incision and a natural pectoral appearance for adolescent patients who have experienced serious emotional problems related to their body images.

Gynecomastia on Thoracic Computed Tomography.

Background and objective Gynecomastia is a benign proliferation of ductal epithelium in the retroareolar region in male patients. The aim of this study was to investigate the frequency of gynecomastia in male patients who underwent thoracic computed tomography (CT) imaging at our clinic, assess possible causes, highlight the imaging characteristics of gynecomastia, and compare our findings with the literature. Materials and methods Male patients over 18 years of age who underwent thoracic CT imaging in our clinic were included in the study. Patients were initially assessed based on age and the presence of gynecomastia. The patients with gynecomastia were evaluated in terms of age, gynecomastia localization (right, left, and bilateral), gynecomastia type (nodular, dentritic, and diffuse), and possible etiology. Results The study included 1500 patients with a mean age of 45.6±21.7 years, and 470 (31.3%) patients had gynecomastia. Gynecomastia was on the right side in 11.3%, on the left side in 11.1%, and bilateral in 77.7% of the patients. Gynecomastia was nodular in 52.1%, dendritic in 35.3%, and diffuse in 17.2% of the patients. The causative factor could not be identified in 44.3% of the patients with gynecomastia. Among cases where the etiology was identified (56.7%), the most common factors were cancer (23.4%), chronic kidney disease (CKD) (13.2%), and chronic hepatitis B (10.7%). Conclusion When evaluating thoracic CT, the breast area, in addition to the lungs, chest wall, and bone structures, should also be evaluated carefully. With the increased use of thoracic CT scans, incidentally detected gynecomastia in patients is also on the rise. Knowing the presence of gynecomastia is very important for the clinician to determine the etiology and treat the underlying disease. Therefore, detecting and reporting gynecomastia on thoracic CT can prevent unnecessary advanced breast imaging methods and play a very important role in treating the underlying etiology.

An Unusual Case of Gynecomastia Associated With Subclinical Hyperthyroidism.

Gynecomastia in males is a medical condition that manifests as the abnormal enlargement of male breast tissue and has a variety of potential causes, which mainly classify as physiologic (infancy, puberty, elderly) and pathologic (hyperthyroidism, medications, cirrhosis, chronic kidney disease (CKD), malignancies). Pathologic causes mainly result from hormonal imbalances. While gynecomastia has been documented in cases of Graves' disease, it is rarely the presenting symptom with very few cases reported in the literature. Here we report an uncommon case of a 65-year-old male with bilateral gynecomastia who presented to his primary care physician (PCP) with concern for breast sensitivity and enlargement. Ultrasound of his breasts showed bilateral findings consistent with gynecomastia. Initial lab demonstrated suppressed thyroid-stimulating hormone (TSH) levels high, normal FT4, elevated estradiol level, elevated sex hormone-binding globulin (SHBG), and elevated total testosterone. The patient was seen by an Endocrinologist six months post-symptom onset and reported that his symptoms had resolved spontaneously.

Comparative analysis of surgical modalities for the gynecomastia treatment: efficiency and aesthetic outcomes.

This retrospective study rigorously compares the clinical efficacy of three surgical methodologies for treating gynecomastia while providing guidance for future surgical modality selection. We analyzed records of 77 gynecomastia patients treated between January 2015 and October 2022. Patients were categorized into three groups: Group A (subcutaneous gland resection via areola incision), Group B (liposuction combined with single-hole endoscopic gland resection), and Group C (liposuction combined with three-hole endoscopic gland resection). Parameters assessed included patient demographics, intraoperative bleeding, surgical duration, hospitalization duration, costs, postoperative drainage, complications, and patient satisfaction. Group A had significantly shorter operation time and lower cost than Groups B and C (P < 0.05). There were no significant differences in postoperative drainage (P > 0.05). Group A had a higher incidence of subcutaneous fluid complications. All groups achieved 100% overall postoperative efficiency. Group B demonstrated superior outcomes for scarring and patient satisfaction. All three surgical modalities effectively treat gynecomastia. Circumareolar incision subcutaneous gland resection is optimal for mild to moderate cases due to reduced operation time and cost. Liposuction with single-hole endoscopic gland resection and three-hole endoscopic gland resection offers fewer complications and discreet incisions. Notably, the liposuction and single-hole endoscopic approach yielded superior postoperative patient satisfaction, aligning with minimally invasive principles and warranting broad clinical application.

[11C]Choline PET/CT in a Patient with Prostate Cancer Biochemical Recurrence Showing Two Suspicious Findings in the Breast and Liver.

A 79-year-old man with prostate cancer (PCa) was referred to our center to perform a [11C]Choline PET/CT for biochemical recurrence. Positron emission tomography/computed tomography (PET/CT) scan detected PCa recurrence in the prostate gland and several pelvic and abdominal lymph nodes. Two abnormal uptakes were also identified in the right breast and in the liver, respectively. Breast histological findings turned out to be gynecomastia, while the liver lesion resulted in a benign perfusion anomaly at follow-up magnetic resonance imaging (MRI). Although incidental findings were benign in this case, it is important to always investigate abnormal uptakes of [11C]Choline, as it could be an expression of further metastases or synchronous malignancies such as breast cancer and hepatocellular carcinoma.

Pseudoangiomatous stromal hyperplasia: a rare cause of gynecomastia in men.

A 17-year-old male with chest malformation and left breast enlargement underwent surgery for gynecomastia. Histological examination revealed mammary fibrous stroma with ductal hyperplasia and features of pseudoangiomatous stromal hyperplasia. Postoperative follow-up showed no complications, but 8 months later, the patient experienced a mild recurrence with enlargement of the nipple-areolar complex. Although recommended for secondary glandular resection, the patient declined further surgery.

Comprehensive aesthetic corrections of gynecomastia using the reproducible safe and minimally invasive surgical strategy.

BACKGROUND: Minimally invasive access and fast recovery are trends of gynecomastia surgery. We placed great importance on liposuction and modified original pull-through technique. The purpose of this study was to present a refined surgical strategy for gynecomastia in grade I and II. METHODS: The refined strategy embraced enhanced liposuction to remove the intraglandular fat sufficiently, followed by open resection of gland using the pull-through and bottom-up technique with adjuvant liposuction in the end. Surgical data were recorded and satisfactory questionnaires with 5-point scales were administered during follow-up. RESULTS: Between January 2017 and May 2022, 165 patients underwent enhanced liposuction combined with the pull-through and bottom-up technique for gland excision. Age ranged from 12 to 56 years. The median length of surgery was 100 min. A median of 300 ml of fat was aspirated and a median of 20.8 g of gland was excised. Seventy-seven patients (46.7%) responded the questionnaires at least 6 months postoperatively, and the average overall satisfaction was 4.68 ± 0.52 points. Thirteen sides of breasts developed complications with a rate of 4.0%. CONCLUSION: Enhanced liposuction combined with pull-through and bottom-up technique proved effective to treat grade I and II gynecomastia with minimal scarring and high satisfaction. The refined strategy was simple and safe, and would obtain optimal outcomes even for inexperienced surgeons.

A Novel Surgical Technique for Gynecomastia: Air-Assisted Minimally Invasive Surgery With Single Axillary Incision.

BACKGROUND: Gynecomastia is a benign condition that develops due to the proliferation of breast tissue in men. Surgical excision is the most effective treatment method. Minimally invasive techniques can be used to avoid visible scarring. We evaluated the efficacy and safety of air-assisted subcutaneous mastectomy in the treatment of gynecomastia. PATIENT AND METHODS: 10 patients with gynecomastia underwent air-assisted subcutaneous mastectomy and liposuction through a single axillary incision, between June 2022 and February 2023. Demographic and clinical data of the patients, duration of surgery, and complications were recorded. The satisfaction levels of the patients regarding physical appearance, mental status, and social environment were measured. The body Q questionnaire was performed preoperatively and in the postoperative third month. RESULTS: The median age was 26 (range, 18-54). Surgical excision was measured as a median of 69 gr (range, 41-177), and liposuction volume was measured as a median of 210 ccs (range, 63-400). The median operation time was 50 minutes (range, 21-60) for excision and 21 minutes (range, 20-75) for liposuction. Body, chest, and nipples related appearance satisfaction levels were measured preoperatively as a median of 44 (range, 36.5-52), 31 (range, 27.5-39), and 51.5 (range, 21-69.8) points vs postoperatively as 92 (range, 92-100), 93 (range, 93-94.8) and 90 (range, 90-100) points, respectively. The patients had a median follow-up of 6 months (range, 3-11). No complications were observed during the follow-up period. CONCLUSION: Air-assisted subcutaneous mastectomy and liposuction is a feasible technique that may provide good cosmetic outcomes by avoiding anterior chest wall scarring.

Primary Anterior Medistinal Choriocarcinoma in Male with Lung Metastasis and Pituitary Microadenoma: A Case Report.

The authors report a case of a 29-year-old male presented with bilateral breast enlargement with no significant past medical history or estrogen exposure. Serum ß-human chorionic gonadotropin (HCG) was 14,306.60 mIU and positron emission tomography-computed tomography discovered a malignant mass on the right side of anterior superior mediastinum. Magnetic resonance imaging demonstrated pituitary microadenoma. Pathological biopsy showed poorly differential pituitary adenoma and immunohistochemical staining displayed that CK(+), PLAP(-), AFP(-), HCG(+), CD30(-), Oct3/4(-), CK7(+), TTF-1(-), CD117(-), Ki 67(80+), CK5/6(-), EMA(partial+), inhibin(partial+). A diagnosis of primary anterior mediastinal choriocarcinoma metastasis to bilateral lungs accompanied with pituitary microadenoma was confirmed. Then the patient received chemotherapy combined with immunotherapy. But serum ß-HCG level was still above the normal, and unfortunately, the patient died 6 months after his diagnosis. This case inspires us to think of the possibility of choriocarcinoma when a man presents gynecomastia or lung metastatic symptoms, adding Opdivo to the chemotherapy might not improve the poor treatment outcomes.