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. 2025 Mar 31;9(13):CASE24769. doi: 10.3171/CASE24769

A neurosurgical approach to skull metastasis from uterine leiomyosarcoma: illustrative case

Arturo Alejandro Carrillo-Uzeta 1,4, Eliany Varela-Avalos 2, Eduardo Agustin-Godinez 3, José Antonio Uehara-Gonzalez 1, José Raymundo Medina-Romero 1, Ana I Velazquez-Zamarripa 3, Alba M Quiñones-González 3, Patricia Anaid Romero-García 2,✉
PMCID: PMC11959634  PMID: 40164004

Abstract

BACKGROUND

Leiomyosarcoma (LMS), a smooth muscle–derived tumor, is associated with a poor prognosis and a high potential for metastasis to local and distal sites. Skull metastases are exceptionally rare, with few cases documented in the literature. Diagnosing and treating these lesions is challenging due to their nonspecific clinical presentation and imaging similarities to other tumors.

OBSERVATIONS

The authors report the case of a 46-year-old female with a 4-month history of a right parietal tumor, accompanied by headaches and left hemiparesis. MRI revealed a heterogeneous, intra- and extra-axial lesion with irregular margins in the right parietal region. Resection of the tumor was performed, and histopathological findings confirmed a diagnosis of metastatic LMS involving the bone and dura. Postoperative radiotherapy was administered, and the patient showed no evidence of recurrence 1 year after initial treatment. A review of the past 30 years of existing case reports of uterine LMS (uLMS) with cranial and intracranial metastases is also provided.

LESSONS

This report highlights the diagnostic and therapeutic complexity of metastatic LMS to the skull. Adjuvant radiotherapy might prove advantageous in improving the prognosis of patients with uLMS, underscoring the significance of integrating clinical, radiological, histopathological, and immunohistochemical findings for accurate diagnosis and management.

https://thejns.org/doi/10.3171/CASE24769

Keywords: metastasis, uterine leiomyosarcoma, skull, neurosurgical oncology, brain, tumor

ABBREVIATIONS: LMS = leiomyosarcoma, uLMS = uterine LMS.

Leiomyosarcoma (LMS) is a smooth muscle–derived tumor mainly found in the retroperitoneum and pelvic area.1 Uterine LMS (uLMS) represents approximately 1% of uterine cancers but is the most common type of uterine sarcoma.2 The incidence of LMS peaks in the 30- to 40-year age range and increases with age, representing 0.2% of uterine tumors at the age of 40 years and reaching up to 1.5% at the age of 70 years.2 The typical clinical presentation of uLMS includes vaginal bleeding, pelvic pain, and a pelvic palpable mass.3 LMS is often incidentally diagnosed through anatomopathological analysis when removing affected tissue such as uteri due to its similarity to common benign uterine fibroids.4 It commonly holds an unfavorable prognosis and high metastasis rates to local and distal sites, including the lungs, liver, pelvic lymph nodes, and vertebral and long bones.5 Metastasis to atypical sites has also been reported; among them, mestastasis to the skull is almost exceptional.5 We describe the case of a 46-year-old postmenopausal female with a single uLMS metastasis to the right parietal bone.

Illustrative Case

Clinical Presentation and Imaging

A 46-year-old postmenopausal woman was referred to the neurosurgery department due to a 4-month history of a painful mass of 16 × 17 cm in the right parietal area accompanied by a constant holocranial headache with a visual analog scale score of 4/10. Thirty days after the onset of her initial symptoms, the patient developed left hemiparesis impairing her daily-life activities and experienced increased headache intensity and episodes of dizziness. As part of her gynecological history, the patient reported a 3-month history of transvaginal bleeding, menopause at the age of 43 years, and an untreated unspecified sexually transmitted disease.

On general examination, the patient was oriented, coherent, and fluent in speech, with a Glasgow Coma Scale score of 15. No alterations were observed in immediate, remote, and long-term memory or in higher-level cognitive skills, gnosis, or praxis. Physical examination revealed a firm, palpable, nonmobile, painful-to-touch, multilobulated mass with irregular edges in the right parietal region. No skin color changes or signs of ulceration were observed. Abdominal examination revealed tenderness with deep palpation and a mobile mass in the lower abdomen, although its exact characteristics could not be entirely determined. The extremities were eutonic and eutrophic, with intact reflexes and a flexor response to the Babinski test. Left hemiparesis was noted, with 3/5 motor strength.

Complementary imaging screenings were requested, and an abdominal CT scan revealed an exophytic, hypodense, infiltrative lesion occupying the entire endometrial cavity, suggesting the primary origin of the tumor (Fig. 1). A contrast-enhanced coronal MR image of the skull and a T1-weighted sequence revealed supratentorial intra- and extra-axial lesions in the right parietal region, with heterogeneous, exophytic, and multilobulated characteristics, along with central hypodensities suggestive of necrosis. A left midline shift of approximately 3 cm was also observed, with perilesional edema and a collapsed right lateral ventricle with dural reinforcement and bone involvement (Fig. 2A and B).

FIG. 1.

FIG. 1.

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Preoperative sagittal abdominal and pelvic CT scan.

FIG. 2.

FIG. 2.

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Preoperative imaging. A: Coronal MR image of the tumor. The orange arrows show intra- and extra-axial tumoral growth. B: MR image of the complete skull. C: Sagittal contrast-enhanced reconstructed CT scan of the skull. D: External sagittal reconstructed CT scan of the skull showing supratentorial intra- and extra-axial tumor in the right parietal bone, neovascularized, with ipsilateral bone destruction.

Contrast-enhanced CT of the skull with 3D reconstruction showed supratentorial intra- and extra-axial lesions in the right parietal region, neovascularized, with ipsilateral bone infiltration and destruction (Fig. 2C and D).

Surgical Findings

After reviewing the imaging findings, a neurosurgical approach was decided, and complete resection of the lesion was performed through a horseshoe incision in the right parietal region, locating and exposing the margins of the lesion and resecting the edges of the bone flap. A complete resection of the intra- and extra-axial lesion was achieved, with posterior duraplasty and placement of a titanium mesh. Intraoperative evaluation revealed an exophytic, multilobulated, right supratentorial, extra-axial lesion measuring 8.5 cm × 7.5 × 7.63 cm, vascularized with invasion of the leptomeninges along with bone destruction of the right parietal bone (Fig. 3).

FIG. 3.

FIG. 3.

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Intraoperative and postoperative photographs. A: Tumor exposure following extra-axial resection, with dural opening revealing intradural invasion. B: Intra-axial exposure and tumor-free margins. C: Resection of lobulated intra-axial lesion. D: Macroscopic photograph of the external surface and the cut surface.

Histological and Immunohistochemical Findings

Histopathology revealed 2 irregularly ovoid tissue structures, the largest with a diameter of 8.5 cm, multilobulated, and grayish-brown in color. On sectioning, the surface was uniformly solid, grayish-white, with yellow areas (Fig. 4A and B). Histological analysis showed a neoplasm characterized by cells with a spindle-shaped and epithelioid appearance, with eosinophilic cytoplasm and poorly defined cell borders. The nuclei were oval and tapered, with irregular nuclear membranes and lumpy chromatin, giving them a vacuolated appearance. Some cells had inconspicuous nucleoli, and abundant atypical mitoses were observed. These cells were arranged in fascicles, whorls, and sheets (Fig. 4C and D). Extensive areas of tumor necrosis were also observed. Based on the morphological appearance, the diagnosis of epithelioid LMS was made.

FIG. 4.

FIG. 4.

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Histopathological findings. A: Macroscopic view of the lesion. B: Tumor sectioning. C and D: Histopathological images revealing a neoplasm characterized by cells with a spindle-shaped and epithelioid appearance, with eosinophilic cytoplasm and poorly defined cell borders. H&E, original magnification ×10 (C) and ×40 (D).

Immunohistochemical studies were performed to confirm the diagnosis using the following markers: 1) estrogen and progesterone receptors, 2) CD10, 3) Pax8, 4) cyclin D1, 5) smooth muscle antigen, and 6) h-caldesmon, of which only the last 2 were diffusely and intensely positive in the cytoplasm of neoplastic cells, thus confirming a diagnosis of LMS. After the histopathological result, the patient was referred for radiotherapy. The surgical approach resolved the patient’s symptoms, and after 1-year of follow-up, the patient was still asymptomatic and without recurrence.

Informed Consent

The necessary informed consent was obtained in this study.

Discussion

Observations

This case report presents a patient with an atypical metastasis to the parietal bone from a uLMS. While not common, a uLMS has the ability to spread to remote areas like the CNS. Brain recurrences have been reported in 20 cases in the last 30 years, and only a few cases involve the skull, as depicted in Table 1. These cases described metastases in locations such as the sphenoid wing; middle fossa and orbit;6 frontal,7 parietal,8 ethmoid, and right orbit;9 and temporo-occipital region.5 In our case, the metastasis was found in the right parietal bone, a rare site, with no other cases reported in this specific location and only a few cases describing metastases to the left parietal area.8,10

TABLE 1.

Summary of cases of brain metastases from LMS reported in literature over the last 30 years

Authors & Year Clinical Presentation Primary Tumor Site Location of Metastases Tx Outcome
Komata et al., 199411 65-yr-old F presented w/ scalp mass uLMS Skull (midfrontal region) Total tumor removal w/ surrounding bone via frontal craniotomy No evidence of recurrence as of July 1993
Wroński et al., 199410 60-yr-old F w/ focal Sz in rt upper extremity & decreased motor function in rt hand; MRI revealed lt parietal lesion uLMS Lt parietal lobe, bilat lung metastases Complete parietal resection of lt parietal metastasis; RT; chemo Died 2.5 yrs after metastasis resection
Uchino et al., 19967 54-yr-old F w/ Hx of uLMS; no neurological Sx on admission uLMS Skull (midfrontal region), multiple distant bone metastases Radical resection of mass via frontal craniotomy Died 2 yrs later of pulmonary recurrences
Ziyal et al., 199912 38-yr-old F w/ lt pain localization, decerebrated on rt, nonverbal, & w/ no eye response to pain stimulus uLMS Dura & frontotemporal brain Total tumor & involved bone removal, rongeured bone, & reconstruction of the dura w/ pericranial graft; RT Died 16 wks after initial neurosurgery
Mawrin et al., 200213 52-yr-old F w/ dysarthria & paresis of rt upper extremity; head MRI showed round isointense lesion in lt pons uLMS Lt pons, bilat lung metastasis VP shunt placement; RT Died approx 2 mos after neurointervention
Sandruck et al., 200414 39-yr-old F w/ heavy menstrual bleeding & pelvic pain uLMS Sphenoid sinus Trans-sphenoidal tumor resection Died 13 mos after intervention
Isobe et al., 200515 74-yr-old M w/ nausea & vomiting; CT revealed cerebellar hemorrhage Duodenum Masses in lt frontal & rt temporal regions; metastases in liver, lt adrenal gland, & duodenum Lt frontal craniotomy, removal of abnormal blood vessels Died on 53rd day after Sx onset
Yip et al., 20068 63-yr-old F w/ 8-cm diameter mass over lt parietal region & progressive rt hemiplegia uLMS Lt occipitoparietal area, involving skull & underlying dura Lt occipitoparietal craniectomy w/ total removal of tumor, & the involved scalp, skull plate, & dura; duraplasty & cranioplasty; skin graft from rt thigh Died approx 4 mos postop
Melone et al., 200816 57-yr-old F w/ mild consciousness impairment; MRI revealed a rt temporopolar mass uLMS Temporopolar lesion, bilat lung lesions Rt temporal craniotomy, complete lesion removal; RT; chemo No data
Honeybul & Ha, 200917 42-yr-old F w/ HA, visual obscuration, & single lt occipital lesion detected on MRI uLMS Lt occipital lesion, multiple pulmonary & peritoneal metastases Image-guided craniotomy & lesion excision Died 2 mos after craniotomy
Yamada et al., 20116 50-yr-old F w/ HA & vomiting, lt hemiparesis & cerebellar ataxia; CT revealed 4-cm subcortical hemorrhage in lt occipital lobe uLMS Multiple lesions in cerebellar vermis, lt occipital lobe, both temporal lobes, rt frontal lobe, & splenium of corpus callosum, lungs, lt femoral head, & rt 4th rib Resection of lt occipital & cerebellar tumors, followed by GKRS of residual tumors Died 12 mos after GKRS
Mariniello et al., 20129 57-yr-old w/ rt proptosis, lt hemiparesis, intracranial hypertension, & torpor uLMS Dura of ant cranial fossa w/ intracranial & intraorbital extension Rt fronto-orbital craniotomy; complete removal of intracranial tumor mass & partial removal of the intraorbital component; chemo Died 13 wks after initial neurosurgery
Inoue et al., 201618 48-yr-old F w/ HA, lt hemiparesis, & aphasia; imaging studies detected solitary brain metastasis in rt frontal lobe uLMS Rt frontal lobe metastasis, pelvic & pulmonary metastases Craniotomy & resection of lesion; chemo Alive 18 mos after brain op
Kim et al., 201619 57-yr-old F w/ protrusion of the lt eye & decreased vision uLMS Lt orbit, lungs, & liver Chemo; pt refused neurointerventions Died 8 wks after initial Dx
Ahuja et al., 201720 60-yr-old F w/ easy fatigability, persistent dull aching HA; presence of 2.5 × 2.5–cm nodule on rt forehead uLMS Rt frontal bone, rt chest wall (abdomen, pleura, 10th rib), both lungs Hysterectomy; chemo; RT; multiple fine needle aspirations from forehead 6-mo FU postchemo, increase of 20% in mass size in lower lobe & rt chest wall
Sosa et al., 201821 43-yr-old F w/ proctalgia & constipation uLMS Meninges Resection through lt frontoparietal craniotomy Died 4 wks after interventions
Juhasz-Böss et al., 20184 30-yr-old F w/ rapidly growing uterus & abnormal bleeding uLMS Pelvic peritoneum & abdominal wall (trocar sites) Complete removal of uterus & resection of metastatic sites during abdominal op No recurrence after 22 mos of FU
Rizzo et al., 20205 39-yr-old F w/ menorrhagia & pelvic pain uLMS Lt pons, bilat lung metastasis VP shunt placement; RT Died approx 2 mos after neurointervention
Imoumby et al., 202122 46-yr-old F w/ rt hemiparesis & HAs uLMS 2 cerebral lesions in lt parieto-occipital region; lung metastases Gross-total resection of brain lesions through classic parietal approach Metastasis recurrence 3 mos postop; died after 2 mos of palliative care
Richards et al., 202323 47-yr-old F w/ persistent HAs, blurry vision, nausea, & vomiting; head CT revealed 4.4-cm mass in rt temporo-occipital region uLMS Lungs & rt temporo-occipital lobe Rt temporo-occipital craniotomy for total tumor resection along w/ adjuvant stereotactic RT & chemo Alive & Sx free 8 mos postresection
Present case 46-yr-old F w/ 16 × 17–cm painful mass rt parietal area, constant holocranial HA, lt hemiparesis, & dizziness uLMS Supratentorial intra- & extra-axial tumor in rt parietal bone Complete resection of intra- & extra-axial lesion, pst duraplasty, & placement of titanium mesh; RT At 1-yr FU, was still alive & free of recurrences
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Ant = anterior; approx = approximately; Dx = diagnosis; FU = follow-up; GKRS = Gamma Knife radiosurgery; HA = headache; Hx = history; pst = posterior; pt = patient; RT = radiotherapy; Sx = symptom(s); Sz = seizure; Tx = treatment; VP = ventriculoperitoneal.

Despite the origin of the primary tumor, metastases to the brain result in nonspecific symptoms like persistent headaches, weakness or numbness, seizures, confusion, or memory loss,24 raising suspicion for differential diagnoses such as meningioma or high-grade gliomas, or in cases of metastasis, metastatic melanoma, endometrial stromal sarcoma, and soft tissue alveolar sarcoma are commonly considered.25,26 Similarly to the aforementioned nonspecific clinical features, our patient presented with a holocranial headache and hemiparesis, signs that are not representative of any specific etiology, posing a diagnostic challenge.

Clinically, there are no specific symptoms associated with uLMS; nonetheless, the most common symptoms include abnormal uterine bleeding, pelvic pain, and a rapidly growing uterine mass.3 In advanced stages, patients might also experience systemic symptoms like fatigue and weight loss.24 In our case, as reported in the literature, our patient (46 years old) falls within the predominant age group seen in this type of tumor as well as experienced some cardinal symptoms such as vaginal bleeding and a uterine mass.

Unfortunately, in our case, the primary tumor was not detected in time due to symptom inconsistency and lack of preventive gynecological control. For that reason, the diagnosis was made after neurosurgical resection, with a suspicion of uterine origin due to preoperative studies. The metastatic tumor was resected entirely, reducing intracranial pressure and improving the patient’s symptoms. A total resection of the tumor was performed, followed by radiotherapy.

Prognosis and Adjuvant Therapies

Overall, the literature describes uLMS prognosis as very poor, with less than 50% of the patients surviving for 5 years or more.27 A study evaluating the survival trends from uterine sarcomas over 10 years in Oxford found that the median overall survival rate, the disease-free survival rate, and progression-free survival in a group of patients with LMS were 25%, 44.5%, and 8 months, respectively, with a 5-year survival rate of 30.5%.28

The current literature suggests that treating uLMS with adjuvant chemotherapy can improve survival rates.23 Typically, chemotherapeutic regimens for advanced LMS involve gemcitabine or doxorubicin.29 A systematic review, meta-analysis, and meta-regression analysis of 51 studies including 1664 patients found that combinations of an anthracycline with an alkylating agent or gemcitabine plus docetaxel yielded the most favorable outcomes when administered as first-line and second-line therapies, respectively.30 Overall, adjuvant chemotherapy has been associated with an average progression-free survival of approximately 5 months.31

The treatment of brain metastases should prioritize neurosurgical resection of any single or multiple recurrences, followed by postoperative chemotherapy.23,32 However, the effectiveness of adjuvant therapy—whether radiotherapy or chemotherapy—in improving survival rates remains inconclusive and limited.16,28,33 As observed in Table 1, it could be implied that survival rates in patients with brain metastases show a worse prognosis in comparison with those patients with localized LMS, ranging from 4 weeks21 up to 2.5 years.10

In contrast to the aforementioned data regarding the current therapeutic guidelines, which suggest chemotherapy as the preferred adjuvant method, it is noteworthy that in our case, the patient received radiotherapy as an adjuvant method without systemic therapy. Subsequent follow-ups revealed that the patient remained recurrence free 1 year after the neurointervention. This highlights the need for future research to standardize adjuvant treatments for uLMS metastatic disease.

Lessons

Worldwide, no reports exist of uLMS metastasizing to the right parietal bone following resection and adjunctive radiotherapy. Our case represents a unique diagnostic and therapeutic challenge. Typically, uLMS metastasis to the CNS is managed with resection and adjuvant chemotherapy or radiotherapy. However, there is an urgent need for additional clinical trials to evaluate the outcomes of adjuvant therapies, particularly for distant metastases to rare sites. Moreover, the absence of population and medical awareness, together with late detection, as seen in our case, contributes to a poorer survival prognosis, emphasizing the need for screening methods and larger-scale data collection and evaluation. Gathering clinical data on atypical presentations is essential to identify variations in disease presentation, diagnosis, and treatment, ultimately facilitating the development of specialized clinical guidelines to improve patient prognosis of this rare and unpredictable disease. Finally, our case underscores the essential role of a multidisciplinary team in managing diseases with a poor prognosis, from early detection to remission.

Acknowledgments

We extend our gratitude to the patient for authorizing the publication of her clinical case for academic and scientific dissemination purposes.

Disclosures

The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

Author Contributions

Conception and design: Romero-García, Carrillo-Uzeta, Varela-Avalos, Agustin-Godinez. Acquisition of data: Romero-García, Carrillo-Uzeta, Agustin-Godinez, Uehara-Gonzalez, Quiñones-González. Analysis and interpretation of data: Romero-García, Carrillo-Uzeta, Varela-Avalos, Agustin-Godinez, Quiñones-González. Drafting the article: Romero-García, Carrillo-Uzeta, Varela-Avalos, Agustin-Godinez, Quiñones-González. Critically revising the article: Romero-García, Carrillo-Uzeta, Varela-Avalos, Medina-Romero, Velazquez-Zamarripa. Reviewed submitted version of manuscript: Romero-García, Carrillo-Uzeta, Varela-Avalos, Velazquez-Zamarripa, Quiñones-González. Approved the final version of the manuscript on behalf of all authors: Romero-García. Statistical analysis: Romero-García, Carrillo-Uzeta. Administrative/technical/material support: Romero-García, Carrillo-Uzeta, Uehara-Gonzalez, Velazquez-Zamarripa. Study supervision: Romero-García, Carrillo-Uzeta, Velazquez-Zamarripa.

Correspondence

Patricia Anaid Romero-García: School of Medicine, Universidad Autónoma de Guadalajara, Zapopan, Jalisco, Mexico. patricia.romero@edu.uag.mx.

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