RESUMO
Primary hyperparathyroidism (PHPT) due to ectopic parathyroid adenoma is a rare case of hypercalcemia in the pediatric population. Herein, a rare case of PHPT due to ectopic intrathymic parathyroid adenoma was described in an asymptomatic 15-year-old girl who had incidental diagnosis based on laboratory abnormalities but experienced a 3-month postoperative course of persistently elevated parathyroid hormone (PTH) and hypercalcemia following the initial unsuccessful parathyroidectomy operation carried out in a non-parathyroid expert center. The curative surgical treatment was accomplished only after the patient was reoperated with video-assisted thoracoscopic surgery (VATS) thymectomy by the surgeon experienced in parathyroid surgery with implementation of the combined imaging modalities for accurate localization of ectopic adenoma including 99mTc sestamibi (MIBI) plus neck and thoracic computed tomography (CT) and the appropriate surgical strategies including intraoperative intact PTH monitoring and frozen section diagnosis. Before the reoperation (VATS thymectomy), laboratory findings showed elevated PTH (1,171 ng/L; reference range: 21.80 ng/L-87.5 ng/L) and hypercalcemia (13.4 mg/dL; reference range: 8.4 mg/dL-10.2 mg/dL). The preoperative PTH levels were 94 ng/L at 5 min after thymectomy and 78 ng/L at 10 min. The PTH and calcium levels were 54.3 ng/L and 8.47 mg/dL, respectively, on postoperative day 1 and were 34.2 ng/L and 8.1 mg/dL on postoperative day 2. The patient was discharged on postoperative day 2 without any complications. In conclusion, our findings indicate the likelihood of isolated primary hyperparathyroidism to be incidentally diagnosed based solely on laboratory abnormalities with no specific clinical manifestations in the pediatric age. In addition, using combined imaging modalities (such as MIBI and CT) in accurate localization of ectopic parathyroid adenoma and implementation of surgery by experienced surgeons along with intraoperative intact PTH monitoring and frozen section diagnosis seem crucial to ensure the curative surgical treatment.
Assuntos
Adenoma , Hiperparatireoidismo Primário , Achados Incidentais , Neoplasias das Paratireoides , Humanos , Feminino , Adolescente , Hiperparatireoidismo Primário/cirurgia , Hiperparatireoidismo Primário/diagnóstico , Hiperparatireoidismo Primário/complicações , Neoplasias das Paratireoides/cirurgia , Neoplasias das Paratireoides/complicações , Neoplasias das Paratireoides/diagnóstico , Neoplasias das Paratireoides/patologia , Adenoma/cirurgia , Adenoma/complicações , Adenoma/diagnóstico , Adenoma/patologia , Paratireoidectomia , Timectomia , Cirurgia Torácica VídeoassistidaRESUMO
BACKGROUND: Complete endoscopic resection of superficial non-ampullary duodenal epithelial tumors (SNADETs) is technically difficult, especially with an extremely high risk of adverse event (AE), although various endoscopic resection methods including endoscopic mucosal resection (EMR), underwater EMR (UEMR), and endoscopic submucosal dissection (ESD) have been tried for SNADETs. Accordingly, a novel simple resection method that can completely resect tumors with a low risk of AEs should be developed. AIMS: A resection method of Noninjecting Resection using Bipolar Soft coagulation mode (NIRBS) which has been reported to be effective and safe for colorectal lesions is adapted for SNADETs. In this study we evaluated its effectiveness, safety, and simplicity for SNADETs measuring ≤ 20 mm. RESULTS: This study included 13 patients with resected lesions with a mean size of 7.8 (range: 3-15) mm. The pathological distributions of the lesions were as follows: adenomas, 77% (n = 10) and benign and non-adenomatous lesions, 23% (n = 3). The en bloc and R0 resection rate was 100% (n = 13). The median procedure duration was 68 s (32-105). None of the patients presented with major AEs including bleeding and perforation. CONCLUSIONS: Large studies such as prospective, randomized, and controlled trials should be conducted for the purpose of validating effectiveness, safety, and simplicity of the NIRBS for SNADETs measuring ≤ 20 mm suggested in this study.
Assuntos
Neoplasias Duodenais , Ressecção Endoscópica de Mucosa , Humanos , Projetos Piloto , Neoplasias Duodenais/cirurgia , Neoplasias Duodenais/patologia , Pessoa de Meia-Idade , Feminino , Masculino , Idoso , Ressecção Endoscópica de Mucosa/métodos , Ressecção Endoscópica de Mucosa/efeitos adversos , Adenoma/cirurgia , Adenoma/patologia , Idoso de 80 Anos ou mais , Eletrocoagulação/métodos , AdultoRESUMO
BACKGROUND: While statistically rare in comparison to other head and neck tumours, parotid gland swellings are often encountered in clinical practice where one of the primary goals of examination becomes distinction between benign and malignant lesions. Hallmarks of malignancy are characterized by a female preponderance, history of radiation exposure, a positive family history, and clinical features like heterogenous consistency, fixity to skin/underlying tissues and involvement of facial nerve. CASE PRESENTATION: Here we present a case of parotid swelling in a 72-year old gentleman from south India that had a curious amalgamation of both benign and malignant features. CONCLUSIONS: While benign, the risk of malignant transformation and rare multicentric occurrence indicates a need to keep basal cell adenoma in mind in case of parotid swellings and their surgical management.
Assuntos
Adenoma , Neoplasias Parotídeas , Humanos , Idoso , Masculino , Adenoma/patologia , Adenoma/diagnóstico por imagem , Adenoma/cirurgia , Adenoma/diagnóstico , Neoplasias Parotídeas/patologia , Neoplasias Parotídeas/diagnóstico , Neoplasias Parotídeas/diagnóstico por imagem , Neoplasias Cutâneas/patologia , Neoplasias Cutâneas/diagnóstico , Neoplasias Cutâneas/cirurgia , Glândula Parótida/patologia , Glândula Parótida/diagnóstico por imagem , Diagnóstico DiferencialAssuntos
Acromegalia , Humanos , Feminino , Acromegalia/etiologia , Acromegalia/tratamento farmacológico , Pessoa de Meia-Idade , Qualidade de Vida , Somatostatina/análogos & derivados , Somatostatina/uso terapêutico , Neoplasias Hipofisárias/complicações , Neoplasias Hipofisárias/cirurgia , Adenoma/complicações , Adenoma/cirurgia , Adenoma/metabolismo , Adenoma/tratamento farmacológico , Agonistas de Dopamina/uso terapêutico , Hormônio do Crescimento Humano , Adenoma Hipofisário Secretor de Hormônio do Crescimento/complicações , Adenoma Hipofisário Secretor de Hormônio do Crescimento/cirurgiaAssuntos
Adenoma , Neoplasias Pulmonares , Humanos , Masculino , Feminino , Adenoma/patologia , Adenoma/genética , Adenoma/cirurgia , Estudos Retrospectivos , Neoplasias Pulmonares/patologia , Neoplasias Pulmonares/genética , Neoplasias Pulmonares/cirurgia , Pessoa de Meia-Idade , Receptores ErbB/genética , Receptores ErbB/metabolismo , MutaçãoRESUMO
Ectopic parathyroid adenoma is a rare pathology. We present a clinical case of a giant ectopic parathyroid adenoma (PA) in an unusual location, which brought significant diagnostical and therapeutical challenges. The tumour in the mediastinum was found incidentally on chest computed tomography (CT). A biopsy was conducted, and histological examination revealed a well-differentiated neuroendocrine tumour. The tumour was excised via right thoracotomy. The final histological examination revealed a parathyroid adenoma, which was unexpected for our team. After three years of observation, there is no evidence of tumour recurrence.
Assuntos
Adenoma , Neoplasias das Paratireoides , Humanos , Neoplasias das Paratireoides/cirurgia , Neoplasias das Paratireoides/diagnóstico por imagem , Neoplasias das Paratireoides/patologia , Adenoma/cirurgia , Adenoma/diagnóstico por imagem , Adenoma/patologia , Neoplasias do Mediastino/cirurgia , Neoplasias do Mediastino/diagnóstico por imagem , Neoplasias do Mediastino/diagnóstico , Neoplasias do Mediastino/patologia , Mediastino/diagnóstico por imagem , Mediastino/patologia , Feminino , Tomografia Computadorizada por Raios X , Masculino , Pessoa de Meia-IdadeRESUMO
Not required for Clinical Vignette.
Assuntos
Neoplasias Hipofisárias , Tomografia de Coerência Óptica , Humanos , Neoplasias Hipofisárias/cirurgia , Neoplasias Hipofisárias/diagnóstico por imagem , Adenoma/cirurgia , Adenoma/diagnóstico por imagem , Eletrorretinografia , Feminino , Células Ganglionares da Retina/patologia , Masculino , Pessoa de Meia-Idade , AdultoRESUMO
BACKGROUND: Duodenal papillary adenoma, a potentially malignant benign tumor is primarily treated with endoscopic papillectomy. Despite its efficacy, endoscopic papillectomy has a high complication rate. This study investigates whether pancreatic duct and common bile duct stent placement can mitigate these complications. METHODS: In a retrospective observational analysis, 79 patients with duodenal papillary adenoma, treated with endoscopic papillectomy at our center, were studied. The cohort included patients who underwent endoscopic papillectomy with no stents placement, common bile duct stent placement alone, pancreatic duct stent placement alone, or stents placement in both ducts. We assessed the outcomes of endoscopic papillectomy, including complete resection rate and recurrence rate as the primary and secondary outcomes respectively. In the meantime, the incidence of complications were also analysed as the safety outcomes. RESULTS: Complete resection rates did not significantly differ between patients with or without stent placement (85.7% P group vs. 89.2% N-P group, P = 0.64). Early complication rates were similar across groups. However, significant reduction in common bile duct stenosis was observed in the stenting group (0% B group vs. 10.5% N-B group, P = 0.03). Furthermore, stent placement correlated with lower adenoma recurrence rates during follow-up (2.4% P group vs. 16.2% N-P group, P = 0.03; 2.4% B group vs. 15.8% N-B group, P = 0.04). CONCLUSIONS: Pancreatic duct and common bile duct stent placement in endoscopic papillectomy may decrease late complications, particularly common bile duct stenosis, and reduce the recurrence of duodenal papillary adenoma. TRIAL REGISTRATION: This study received approval from the Institutional Review Board and Ethics Committee of Beijing Friendship Hospital (Approval No. BFHHZS20230203), and retrospectively registered in www. CLINICALTRIALS: gov (NCT06301048, Initial Release date: 02/18/2024, Last Public Release date: 03/03/2024).
Assuntos
Adenoma , Neoplasias Duodenais , Ductos Pancreáticos , Stents , Humanos , Masculino , Estudos Retrospectivos , Feminino , Pessoa de Meia-Idade , Adenoma/cirurgia , Adenoma/patologia , Neoplasias Duodenais/cirurgia , Neoplasias Duodenais/patologia , Ductos Pancreáticos/cirurgia , Ductos Pancreáticos/patologia , Idoso , Complicações Pós-Operatórias/etiologia , Complicações Pós-Operatórias/epidemiologia , Resultado do Tratamento , Recidiva Local de Neoplasia , Esfinterotomia Endoscópica/métodos , Esfinterotomia Endoscópica/efeitos adversos , Adulto , Ampola Hepatopancreática/cirurgia , Ducto Colédoco/cirurgiaRESUMO
In this video, we demonstrate the technique of endoscopic transpterygoid trans lacerum trans cavernous approach for the excision of Knosp 4 functioning pituitary macroadenomas. We highlight the anatomy and key steps of the approach using cadaveric dissection and present two clinical cases. A 42-year-old female with a growth hormone-secreting tumor and Knosp 4 macroadenoma underwent an extended endoscopic endonasal approach and near-total excision of the tumor. Postoperatively she was neurologically intact. Postoperative MR showed a small residue in the lateral compartment of the cavernous sinus and along the right optic tract superiorly. Due to the proximity of the residual tumor near the optic apparatus, she was offered a pterional approach, and the tumor around the optic apparatus was excised. She was given stereotactic radiosurgery for the small residual tumor in the cavernous sinus and is currently doing well, under remission. The second case was a 23-year young male with acromegaly and bilateral Knosp 4 macroadenoma. He underwent endoscopic transpterygoid trans lacerum trans cavernous approach and gross total excision of the tumor was done. He had transient bilateral ophthalmoplegia in the immediate postoperative period which recovered fully within 6 weeks. He is currently in remission without any need for radiotherapy. To conclude, an in-depth knowledge of the anatomy helps in improving surgical outcomes in extended endoscopic endonasal surgeries.
Assuntos
Adenoma , Seio Cavernoso , Neuroendoscopia , Neoplasias Hipofisárias , Humanos , Adulto , Feminino , Neoplasias Hipofisárias/cirurgia , Neoplasias Hipofisárias/diagnóstico por imagem , Adenoma/cirurgia , Adenoma/diagnóstico por imagem , Masculino , Seio Cavernoso/cirurgia , Seio Cavernoso/diagnóstico por imagem , Neuroendoscopia/métodos , Adulto JovemRESUMO
BACKGROUND: Parathyroid gland (PG) surgery is often challenging due to the small size and indistinct nature of these glands. The introduction of intraoperative near-infrared autofluorescence (NIRAF) has shown promise in localizing parathyroid tissue. However, the NIRAF features of parathyroid adenomas remain unclear. The aim of this study is to assess the NIRAF pattern of parathyroid adenomas. METHODS: Patients who underwent surgery for primary hyperparathyroidism at the University Hospital of Pisa, Endocrine Surgery Unit, between December 2021 and February 2022 were enrolled in this study. Intraoperative NIRAF patterns of suspected parathyroid adenomas were evaluated, with particular attention given to the presence of a bright cap. RESULTS: A retrospective study was conducted on 11 patients with primary hyperparathyroidism who underwent parathyroidectomy at our institution. Histopathological examination of the 15 resected specimens confirmed 14 parathyroid adenomas (12 chief cell parathyroid adenomas, 1 oxyphil cell parathyroid adenoma, and 1 mixed cell parathyroid adenoma) and one schwannoma. All adenomas exhibited a heterogeneous NIRAF pattern, distinct from the homogeneous pattern observed in the schwannoma. A bright cap was identified in 9 out of 14 (64.3%) parathyroid adenomas (all chief cell adenomas). On the contrary, all 9 macroscopically normal PGs identified during surgery presented an homogeneous pattern. CONCLUSION: Our findings support the integration of NIRAF into parathyroid surgical procedures. The heterogeneous NIRAF pattern observed in parathyroid adenomas, often accompanied by a bright cap, offers a promising intraoperative diagnostic tool to differentiate hyperfunctioning from normal parathyroid tissue. Larger-scale randomized trials are warranted to further validate these findings.
Assuntos
Adenoma , Imagem Óptica , Neoplasias das Paratireoides , Paratireoidectomia , Humanos , Neoplasias das Paratireoides/patologia , Neoplasias das Paratireoides/cirurgia , Neoplasias das Paratireoides/diagnóstico por imagem , Feminino , Masculino , Estudos Retrospectivos , Pessoa de Meia-Idade , Adenoma/patologia , Adenoma/cirurgia , Adenoma/diagnóstico por imagem , Paratireoidectomia/métodos , Idoso , Imagem Óptica/métodos , Adulto , Hiperparatireoidismo Primário/cirurgia , Hiperparatireoidismo Primário/patologia , Glândulas Paratireoides/patologia , Glândulas Paratireoides/diagnóstico por imagem , Glândulas Paratireoides/cirurgia , Espectroscopia de Luz Próxima ao Infravermelho/métodosRESUMO
Introduction: Pituitary adenomas (PAs) are benign tumors with high prevalence and, occasionally, aggressive course. The tumorigenesis of these lesions is not completely understood at the molecular level. BAK1 and BAX proteins play fundamental roles in apoptosis and seem to interact with VDAC proteins, whose expressions have been markedly altered in cancer, impacting their prognosis. Objective: to evaluate the gene expression of VDAC1, VDAC2, BAK1 and BAX and their association with clinical and imaging characteristics in PA. Methods: Clinical-epidemiological data were collected from 117 tumor samples from patients affected by PA. Invasiveness was assessed by the Knosp scale. Gene expression was examined by real-time PCR. Relative expression analysis was performed by 2^(-DDCt) method. Results: The sample was mainly composed of women (69/117 - 57.2%). Tumor subtypes observed were Non-Functioning (NF) (73/117 - 62.4%), Acromegaly (24/117 - 20.5%) and Cushing's Disease (CD) (20/117 - 17.1%). Compared to normal tissue, there was a significant reduction in VDAC1 expression in the Acromegaly (p=0.029) and NF (p=0.002) groups. BAX expression was lower in all groups (p <0.001; p=0.007; P =0.005). No difference was found in VDAC2 and BAK1 expression, compared to normal pituitary. Overexpression of VDAC2 occurred in PAs with post-surgical regrowth (p=0.042). A strongly negative correlation was observed in BAX and BAK1 expression in CD. Conclusion: The results indicated that downregulations of VDAC1 and BAX may be related to resistance to apoptosis. In contrast, overexpression of VDAC2 in regrowing PAs suggests an antiapoptotic role for this gene. In summary, the genes evaluated might be involved in the biopathology of PAs.
Assuntos
Adenoma , Neoplasias Hipofisárias , Canal de Ânion 1 Dependente de Voltagem , Proteína Killer-Antagonista Homóloga a bcl-2 , Humanos , Neoplasias Hipofisárias/genética , Neoplasias Hipofisárias/metabolismo , Neoplasias Hipofisárias/cirurgia , Neoplasias Hipofisárias/patologia , Feminino , Adenoma/genética , Adenoma/metabolismo , Adenoma/cirurgia , Adenoma/patologia , Pessoa de Meia-Idade , Masculino , Adulto , Proteína Killer-Antagonista Homóloga a bcl-2/genética , Proteína Killer-Antagonista Homóloga a bcl-2/metabolismo , Canal de Ânion 1 Dependente de Voltagem/genética , Canal de Ânion 1 Dependente de Voltagem/metabolismo , Canal de Ânion 2 Dependente de Voltagem/genética , Canal de Ânion 2 Dependente de Voltagem/metabolismo , Proteína X Associada a bcl-2/genética , Proteína X Associada a bcl-2/metabolismo , Prognóstico , Idoso , Regulação Neoplásica da Expressão Gênica , Biomarcadores Tumorais/genética , Biomarcadores Tumorais/metabolismo , Proteínas Proto-Oncogênicas c-bcl-2/genética , Proteínas Proto-Oncogênicas c-bcl-2/metabolismoRESUMO
BACKGROUND: Functional pituitary adenomas (PAs) manifest as intricate clinical syndromes, and surgery emerges as the principal intervention to mitigate associated morbidity and mortality. The endoscopic transsphenoidal surgery (ETS) approach stands as the preferred method for addressing PAs, with postoperative remission acting as a pivotal prognostic factor. METHODS: This study seeks to evaluate the influence of different surgical techniques and complications of ETS on functional PAs, focusing on both Acromegaly and Cushing's disease (CD). Patient records, including characteristics, perioperative assessments, postoperative complications, and follow-up data, were systematically gathered. Tumor resection methods were categorized into: (I) complete pseudocapsule resection; and (II) complete piecemeal resection. Post-surgery, daily monitoring of serum cortisol levels continued for a consecutive 3-day period until values of 2 µg/dL or less were achieved. Growth hormone levels were reassessed 12 weeks later, targeting a level of <0.14 g/L. A follow-up enhanced magnetic resonance imaging examination was conducted 3 months post-surgery to confirm the absence of residual tumors. RESULTS: The study identified 46 patients (23 with acromegaly and 13 with CD) who underwent endoscopic surgery between 2020 and 2023. Twenty-six patients underwent piecemeal resection (January 2020 to December 2022), while 10 patients underwent complete pseudocapsule removal (January to December 2023). No significant changes in surgical complications were observed between the two techniques. No instances of carotid artery injury, epistaxis, intracranial infection, or loss of olfaction were reported. In the pseudocapsule group, one patient experienced transient vision loss. Notably, 80% of patients in the pseudocapsule group achieved remission as compared to 57.7% in the piecemeal group. CONCLUSIONS: Pseudocapsule-based resection exhibited a remarkable remission rate, a low complication rate, and an absence of recurrence in functional adenoma patients. Despite the limited number of cases and our early experience, further studies are warranted to validate its effectiveness and safety.
Assuntos
Neoplasias Hipofisárias , Humanos , Masculino , Feminino , Neoplasias Hipofisárias/cirurgia , Pessoa de Meia-Idade , Adulto , Endoscopia/métodos , Adenoma/cirurgia , IdosoRESUMO
BACKGROUND: Endonasal endoscopic approach (EEA) has become an integral part of neurosurgery, particularly for managing various pathologies involving the sellar and parasellar region and removing lesion in that area. But there is a rare condition in this area that not so common in literature is pituitary abscess (PA) after EEA for pituitary adenoma. CASE DESCRIPTION: A healthy 57-year-old man presented with a recurrent of bitemporal hemianopsia, persistent hypoadrenocorticism. In the history, he underwent two times of endonasal endoscopic transphenoid to remove pituitary adenoma. Magnetic resonance imaging (MRI) showed a sphenoid and sellar lesion with suprasellar extension and compression of the optic chiasm, with homogenous signal inside. Endoscopic transphenoid surgery was performed and the lesion was found is an abscess inside sphenoid and sellar with frank pus was obtained. But culture of the fluid was negative. After the operation, the patient was treated with antibiotics. His symptoms resolved after EEA, he improved visual field at this time but still in hypoadrenocorticism condition. An MRI was obtained after the operation to reveal that the lesion in sphenoid and sellar totally removed. We report on a rare case of a PA after two times of endonasal endoscopic transphenoid surgery of pituitary adenoma, and discuss the management of these conditions. We report a patient that came with us with recurrent of bitemporal hemianopsia and persistent hypoadrenocorticism. The patient undergone endonasal endoscopic sphenoid two times in the past. He didn't have clinical signs of infection. Imaging feature on MRI was high T2 and also high on T1 with homogenous signal inside with size 3.2 cm × 3.2 cm. Another EEA for this patient was performed to remove abscess and obtain pus from the lesion, but the result of culture was negative. He improved visual field but still in hypoadrenocorticism. CONCLUSIONS: In this case we report on a rare complication of transphenoid surgery, a PA progresses after EEA for a pituitary tumor. With this clinical experience, the correct diagnosis of PA before surgery, so it is a key leading to an exactly treatment for this condition. endonasal endoscopic transphenoid approach to drainage and removing abscess, and using antibiotics are necessary to improve the outcome of PA.
Assuntos
Neoplasias Hipofisárias , Humanos , Masculino , Pessoa de Meia-Idade , Neoplasias Hipofisárias/cirurgia , Neoplasias Hipofisárias/complicações , Adenoma/cirurgia , Adenoma/complicações , Endoscopia/métodos , Imageamento por Ressonância Magnética/métodos , Doenças da Hipófise/cirurgia , Abscesso Encefálico/cirurgia , Abscesso Encefálico/etiologiaRESUMO
BACKGROUND: Sessile serrated lesions (SSL) account for up to 30% of colorectal carcinoma pathogenesis. With multiple classification changes and improvements in colonoscopy equipment and technique, historical reporting may have underestimated the true incidence of SSLs. This study aimed to determine the incidence of SSLs in patients undergoing colonoscopic investigation in Canterbury, New Zealand over a 1-year period and describe their clinical and pathological characteristics. METHODS: Electronic records were searched to identify all lower endoscopy procedures with polypectomy performed from 1 January 2022 to 1 December 2022 (inclusive). Patients' electronic records were used to collect histological classification, location and size of each polyp removed during their procedure. The primary outcome was the number of procedures that had one or more SSL, adenoma or hyperplastic polyp identified. Secondary outcomes included histological classification, location and size of each polyp removed. RESULTS: There were 4346 procedures completed during the study period. Of these, 64.1% (2786) had a polypectomy and 18.6% (808) had at least one SSL excised. Individual polyp analysis was completed on 9166 polyps and found that 24.0% of polyps removed were SSLs and they were found predominately in the right colon (65.1% right colon, 32.6% left colon, 2.3% rectum). SSLs were typically <10 mm (84.8%). CONCLUSION: This study found a higher incidence of SSLs compared to previous research. These results raise questions regarding whether SLL rates have been historically underestimated, whether SSL detection rate should be included as a key performance indicator and raises further concerns regarding the use of computed tomography colonography as a screening tool.
Assuntos
Pólipos do Colo , Colonoscopia , Neoplasias Colorretais , Humanos , Incidência , Pólipos do Colo/patologia , Pólipos do Colo/cirurgia , Pólipos do Colo/epidemiologia , Masculino , Feminino , Nova Zelândia/epidemiologia , Pessoa de Meia-Idade , Idoso , Neoplasias Colorretais/patologia , Neoplasias Colorretais/epidemiologia , Neoplasias Colorretais/cirurgia , Adenoma/patologia , Adenoma/epidemiologia , Adenoma/cirurgia , Estudos RetrospectivosRESUMO
BACKGROUND AND OBJECTIVE: Cushing disease (CD) affects mortality and quality of life along with limited long-term remission, underscoring the need to better identify recurrence risk. The identification of surgical or imaging predictors for CD remission after transsphenoidal surgery has yielded some inconsistent results and has been limited by single-center, single-surgeon, or meta-analyses studies. We sought to evaluate the multicenter Registry of Adenomas of the Pituitary and Related Disorders (RAPID) database of academic US pituitary centers to assess whether robust nonhormonal recurrence predictors could be elucidated. METHODS: Patients with treated CD from 2011 to 2023 were included. The perioperative and long-term characteristics of CD patients with and without recurrence were assessed using univariable and multivariable analyses. RESULTS: Of 383 patients with CD from 26 surgeons achieving postoperative remission, 288 (75.2%) maintained remission at last follow-up while 95 (24.8%) showed recurrence (median time to recurrence 9.99 ± 1.34 years). Patients with recurrence required longer postoperative hospital stays (5 ± 3 vs 4 ± 2 days, P = .002), had larger average tumor volumes (1.76 ± 2.53 cm 3 vs 0.49 ± 1.17 cm 3 , P = .0001), and more often previously failed prior treatment (31.1% vs 14.9%, P = .001) mostly being prior surgery. Multivariable hazard prediction models for tumor recurrence found younger age (odds ratio [OR] = 0.95, P = .002) and Knosp grade of 0 (OR = 0.09, reference Knosp grade 4, P = .03) to be protective against recurrence. Comparison of Knosp grade 0 to 2 vs 3 to 4 showed that lower grades had reduced risk of recurrence (OR = 0.27, P = .04). Other factors such as length of stay, surgeon experience, prior tumor treatment, and Knosp grades 1, 2, or 3 failed to reach levels of statistical significance in multivariable analysis. CONCLUSION: This multicenter study centers suggests that the strongest predictors of recurrence include tumor size/invasion and age. This insight can help with patient counseling and prognostication. Long-term follow-up is necessary for patients, and early treatment of small tumors may improve outcomes.
Assuntos
Hipersecreção Hipofisária de ACTH , Sistema de Registros , Humanos , Hipersecreção Hipofisária de ACTH/cirurgia , Masculino , Feminino , Pessoa de Meia-Idade , Adulto , Resultado do Tratamento , Recidiva Local de Neoplasia/epidemiologia , Recidiva Local de Neoplasia/prevenção & controle , Indução de Remissão , Adenoma/cirurgiaRESUMO
RATIONALE: Acromegaly, predominantly resulting from a pituitary adenoma, is marked by excessive secretion of growth hormone (GH) and insulin-like growth factor-1 (IGF-1). However, normalization of blood glucose levels posttreatment is rarely achieved. This case study aims to highlight the diagnostic challenges posed by overlapping symptoms of acromegaly and diabetes, emphasizing the importance of precise diagnosis and effective treatment strategies for optimal patient outcomes. PATIENT CONCERNS: A 22-year-old male was hospitalized for diabetic ketoacidosis and exhibited classic signs of acromegaly, such as enlarged hands and feet, and distinct facial changes. DIAGNOSES: The patient's diagnosis of acromegaly, attributed to a pituitary adenoma, was confirmed through clinical observations, laboratory findings (notably raised serum GH and IGF-1 levels, and absence of GH suppression after glucose load during an OGTT), and pituitary MRI scans. INTERVENTIONS: The patient underwent 2 surgical tumor resections followed by gamma knife radiosurgery (GKRS). After treatment, GH, IGF-1, and blood glucose levels normalized without further need for hypoglycemic intervention. OUTCOMES: Posttreatment, the patient achieved stable GH, IGF-1, and blood glucose levels. The hyperglycemia was attributed to the GH-secreting tumor, and its resolution followed the tumor's removal. LESSONS: This case emphasizes the need for comprehensive assessment in patients with acromegaly to address coexisting diabetic complications. Surgical and radiotherapeutic management of acromegaly can lead to significant metabolic improvements, highlighting the importance of interdisciplinary care in managing these complex cases.
Assuntos
Acromegalia , Humanos , Masculino , Acromegalia/etiologia , Acromegalia/diagnóstico , Acromegalia/complicações , Acromegalia/terapia , Adulto Jovem , Fator de Crescimento Insulin-Like I/metabolismo , Fator de Crescimento Insulin-Like I/análise , Neoplasias Hipofisárias/complicações , Neoplasias Hipofisárias/cirurgia , Neoplasias Hipofisárias/diagnóstico , Hormônio do Crescimento Humano/sangue , Adenoma/complicações , Adenoma/cirurgia , Radiocirurgia/métodos , Diabetes Mellitus , Cetoacidose Diabética/complicações , Cetoacidose Diabética/terapia , Cetoacidose Diabética/diagnóstico , Glicemia/análise , Glicemia/metabolismoRESUMO
The patient is a 28-year-old male who presented with hemifield slide for 10 months following surgery for a large pituitary adenoma. A comprehensive ophthalmological examination and visual function tests were conducted, including best corrected visual acuity, visual field, stereopsis, accommodative convergence, Worth four-dot test, and prism cover test for quantifying strabismus, along with assessments using a depression scale and a visual function questionnaire. Rehabilitation plans included applying press-on prisms to correct vertical strabismus, binocular vision training, and saccadic training. After rehabilitation, the patient was able to maintain binocular single vision at near and intermediate distances, experienced improved visual comfort, and showed significant improvements in scores on the depression scale and visual function questionnaire.
Assuntos
Neoplasias Hipofisárias , Humanos , Masculino , Adulto , Neoplasias Hipofisárias/cirurgia , Neoplasias Hipofisárias/reabilitação , Acuidade Visual , Visão Binocular , Estrabismo/cirurgia , Adenoma/cirurgia , Adenoma/reabilitação , Campos VisuaisRESUMO
BACKGROUND: Although the skeleton remains a common target of primary hyperparathyroidism, the classic bone disease "osteitis fibrosa cystica" is currently rare due to early diagnosis. This case represents severe classic bone manifestations of primary hyperparathyroidism due to delayed diagnosis and delayed medical attention. CASE PRESENTATION: A 19-year-old young female was symptomatically managed for chronic back pain and nonspecific bone pain in the small joints of both hands over 2 months by a general practitioner. The patient had delayed seeking for treatment for 3 months. Later, she was evaluated for tuberculosis, hematological malignancies and rheumatic disorders following a fractured T12 vertebra and underwent pedicle screw fixation. However, clinical examination and investigations, including biochemistry, imaging and histology, ruled out the above conditions. Unfortunately, serum calcium level was not performed at the initial presentation. Later, primary hyperparathyroidism was diagnosed on the basis of moderate hypercalcaemia and elevated intact PTH levels (2064 pg/ml). She had sufficient vitamin D levels and normal kidney function. Her DXA scan revealed severe secondary osteoporosis with the lowest Z score of -8 at the total lumbar spine. Ultrasonography of the thyroid revealed a hypo echoic mass in the left lower neck, and localization studies with technetium-99 m sestamibi and 4D-CT revealed a left inferior parathyroid adenoma (1.6 × 1.5 × 1.6 cm). CT scan also revealed brown tumors in the mandible and vertebrae and diffuse bony changes in the skull, sternum, humerus and vertebrae. Her radiographs revealed subperiosteal bone resorption on the radial aspects of the middle and distal phalanges and brown tumors in both the ulna and fibula. We excluded MEN and other hereditary syndromes in our patient with a personal and family history and with a normal pituitary hormone profile because of poor resources for genetic testing. She underwent parathyroid adenoma excision, and the postoperative period was complicated with hungry bone syndrome, requiring high doses of calcium and active vitamin D supplements. These supplements were gradually weaned off over 6 months, and she recovered with normal biochemical investigations. Histology revealed parathyroid adenoma without malignant features. CONCLUSION: In developing countries where routine calcium screening is not available, clinicians should be aware of various manifestations of primary hyperparathyroidism to allow diagnosis as soon as possible without delay to prevent further progression, as it is a treatable condition.
Assuntos
Fraturas da Coluna Vertebral , Humanos , Feminino , Fraturas da Coluna Vertebral/etiologia , Fraturas da Coluna Vertebral/cirurgia , Fraturas da Coluna Vertebral/patologia , Adulto Jovem , Hiperparatireoidismo Primário/cirurgia , Hiperparatireoidismo Primário/complicações , Hiperparatireoidismo Primário/etiologia , Hiperparatireoidismo Primário/patologia , Hiperparatireoidismo Primário/diagnóstico , Neoplasias das Paratireoides/cirurgia , Neoplasias das Paratireoides/complicações , Neoplasias das Paratireoides/patologia , Adolescente , Adenoma/complicações , Adenoma/patologia , Adenoma/cirurgia , Osteíte Fibrosa Cística/etiologia , Osteíte Fibrosa Cística/patologiaRESUMO
This paper aims to analyze the risk factors for the recurrence or progression of non-functioning pituitary adenomas (NFPAs) in male patients after transnasal sphenoidal surgery and to develop a predictive model for prognosis. Clinical and follow-up data of 126 male patients with NFPAs treated by transnasal sphenoidal surgery from January 2011 to January 2021 in Fuzhou 900th Hospital were retrospectively analyzed. Lasso regression analysis was used to screen the best predictors, and the predictors were further screened by multivariate logistic regression analysis, and the nomogram prediction model was constructed. The performance of the model was verified by three aspects: discrimination, calibration and clinical utility by using the consistency index (C-index), receiver operating characteristic curve (ROC), calibration curve, clinical decision curve (DCA) and Clinical impact curve (CIC). Out of 126 cases, 7 (5.56%) showed postoperative tumor recurrence, and 18 (14.29%) exhibited postoperative residual regrowth (progression). Age (P = 0.024), maximum tumor diameter (P < 0.001), modified Knosp grade (P < 0.001), resection extent (P < 0.001), Ki67 (P < 0.001), pressure symptom (P < 0.001), Pre-op hypopituitarism (P = 0.048), Post-op new hypopituitarism (P = 0.017) showed significant differences among the recurrence group, the progression group, and the alleviation group. Three independent risk factors (Ki67, modified Knosp grade, and resection extent) affecting postoperative remission were used to construct a predictive model for long-term postoperative failure to remit. The C-index of the nomogram model was 0.823, suggesting that the model had a high discriminatory power, and the AUC of the area under the ROC curve was 0.9[95% CI (0.843, 0.958)]. A nomogram prediction model based on modified Knosp grading (grades 3B-4), resection extent (partial resection), and Ki-67 (≥ 3%) predicts the recurrence or progression of NFPAs in men after transnasal sphenoidal surgery.