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PubMed1978

Radiologic findings of hamartomas of the tuber cinereum and hypothalamus.

Five cases, four histologically proved, or hamartoma of the tuber cinereum and hypothalamus in children (age range: 2--12 years) are reported. Three cases had pubertas praecox, and in all of these the hamartoma was located in the basal cistern between the chiasm and pons, and had a collar button shape and size typical of hamartoma of the tuber cinereum. In the third case, which presented with headache, a huge calcified mass in the suprasellar region was initially thought to be craniopharyngioma. The fourth case had a hamartoma within the substance of the hypothalamus and presented with hyponatremia and temporal lobe seizures.

Brain Neoplasms↗
PubMed1986

Pulmonary hamartoma: CT findings.

Forty-seven patients with a proved (n = 31) or presumed (n = 16) diagnosis of pulmonary hamartoma were studied prospectively by thin-section computed tomography (CT). CT criteria for hamartoma included a diameter of 2.5 cm or less, a smooth edge, and focal collections of fat or fat alternating with areas of calcification. No case of cancer (n = 283) or metastatic disease (n = 72) fulfilled these criteria. Seventeen hamartomas with no detectable calcium or fat were not diagnosed by means of CT. Two other lesions contained diffuse calcium deposits. In 28 lesions, a CT diagnosis of hamartoma was based on the detection of fat (n = 18) or calcium plus fat (n = 10). Twelve such cases were proved histologically by means of thoracotomy or needle biopsy; the remainder, including eight in asymptomatic patients aged 65 years or older, were managed with conservative follow-up.

Adult↗
PubMed1989

Breast hamartomas: variable mammographic appearance.

The mammograms of 17 women with pathologically proved breast hamartomas were reviewed. Abnormal masses were detected on 12. Nine women had masses with benign features. Two of these had findings considered classic for hamartoma. In three cases, the appearance of the mass was suggestive of carcinoma. The breasts were very dense in four of five women without detectable mass. The findings suggest that the classic mammographic appearance of breast hamartomas is less common than previously reported, which may be explained by earlier detection of small hamartomas.

Adult↗
PubMed1997

Splenic hemangiomas and hamartomas: MR imaging characteristics of 28 lesions.

PURPOSE: To determine the magnetic resonance (MR) imaging features of splenic hemangiomas and hamartomas, including their pattern of dynamic contrast material enhancement. MATERIALS AND METHODS: The appearance of 28 lesions in 18 patients was retrospectively reviewed on T2-weighted images (16 patients), unenhanced T1-weighted images (18 patients), and dynamic contrast-enhanced T1-weighted images (17 patients). Seventeen of 23 hemangiomas and all five hamartomas were proved at pathologic examination. RESULTS: Of the 22 hemangiomas imaged with T2-weighting, 19 were hyperintense, two were isointense, and one was hypointense relative to the spleen. Dynamic gadolinium-enhanced imaging demonstrated a progressive centripetal pattern of enhancement in 19 of 22 hemangiomas. On delayed images, 19 hemangiomas demonstrated uniform enhancement. Of the five hamartomas, four were imaged with T2-weighting; three were hyperintense and one was hypointense relative to the spleen. All hamartomas demonstrated diffuse heterogeneous enhancement on images obtained early after administration of contrast material and became more uniformly enhanced on delayed images. CONCLUSION: Splenic hemangiomas showed signal intensity characteristics and enhancement patterns similar to those described for hepatic hemangiomas. Since these features have been shown to reliably distinguish hemangiomas from other benign and malignant liver lesions, it may be reasonable to consider without histologic verification that lesions in the spleen with these imaging features represent hemangiomas.

Adolescent↗
PubMed2006

Giant hypothalamic hamartoma operated through subfrontal approach with orbitary rim osteotomy.

INTRODUCTION: Hypothalamic hamartomas are associated with precocious puberty, gelastic seizures and severe refractory epilepsy. Treatment options include surgical resection, radiofrequency and radiosurgery. CASE REPORT: A 7-month-old girl presented with gelastic seizures and developmental delay related to a giant hypothalamic hamartoma. The patient was operated through a subfrontal approach. Intraoperatively the lesion appeared intimately adherent to the right internal carotid artery. Seizure control was improved after tumoral decompression. CONCLUSIONS: Treatment of giant hypothalamic hamartomas should always include surgical resection, given the mass effect over surrounding vital structures. Subfrontal approach with orbitary rim osteotomy provides a wide exposure with minimal frontal lobe retraction. Close adherence of hypothalamic hamartoma to vascular structures may be present, requiring careful surgical manipulation.

Craniotomy↗
PubMed1989

Lack of adrenarche in two children with precocious puberty secondary to hypothalamic hamartoma.

Adrenarche, which occurs earlier than gonadarche in normal children, is marked by increases in plasma dehydroepiandrosterone and its sulfate (DHAS). Adrenarche and gonadarche can be dissociated in various situations, e.g. central precocious puberty, indicating that they are controlled by independent mechanisms. This report concerns 2 children with central precocious puberty secondary to hypothalamic hamartoma. Their plasma basal DHAS values, compared to other cases with central precocious puberty not secondary to hamartoma, remained low for chronological age and bone age over a follow-up of 6.3 (case 1) and 9.2 9.2 years (case 2): in case 1 (boy), DHAS was 9 micrograms/dl at chronological age 7.7 and bone age 13 years; in case 2 (girl), DHAS was 11 micrograms/dl for chronological age 10.5 and bone age 13.5 years. GH secretion was normal. Basal plasma cortisol levels as the levels during hypoglycemia and after corticotropin stimulation were all normal. These data suggest that hypothalamic hamartoma may affect the central control of adrenarche. They may also contribute to the diagnosis of hypothalamic hamartoma.

Adrenal Glands↗
PubMed2002

Resection of the lesion in patients with hypothalamic hamartomas and catastrophic epilepsy.

BACKGROUND: Patients with hypothalamic hamartomas (HH) often have severe refractory epilepsy, incapacitating behavioral abnormalities, and cognitive decline. Attempts to control the seizure disorder by resection of apparently epileptogenic mesial temporal or other cortical structures have failed consistently. OBJECTIVE: To report a series of 13 patients in whom the hamartoma itself was resected. METHODS: All patients underwent preoperative evaluation between ages 2 and 33 years and had subtotal or complete resection of the hamartoma. Follow-up ranged from 1 to 5.5 years (mean: 2.8 y). RESULTS: Preoperatively, all patients had variable combinations of gelastic, complex partial, and generalized seizures. Eight had drop attacks. In addition, all had marked behavior abnormalities and cognitive impairment. Postoperatively, two patients are completely seizure-free and 11 are either seizure-free or have achieved a greater than 90% reduction of drop attacks and generalized tonic-clonic seizures. However, minor gelastic, complex partial, and atypical absence seizures have persisted in 11 patients, although at significantly reduced rates. In addition, there has been a dramatic improvement in behavior and cognition. Three patients had an anterior thalamic and one a capsular infarct, which left only minimal long-term deficits. Exact location of the lesion in relation to the interpeduncular fossa and the walls of the third ventricle correlated with extent of excision, seizure control, and complication rate. CONCLUSION: Resection can alleviate both the seizures and the behavioral and cognitive abnormalities of hypothalamic hamartomas, but complications are frequent.

Adolescent↗
PubMed1993

Asymptomatic large hypothalamic hamartoma associated with polydactyly in an adult.

A hypothalamic hamartoma is a congenital tumor-like neural malformation. It is usually seen in children and is associated with neuroendocrinological symptoms, seizures, or psychological impairments. An asymptomatic hypothalamic hamartoma in an adult is extremely rare. This report describes an asymptomatic adult with a large hypothalamic hamartoma associated with polydactyly in his feet. Both polydactyly and hamartoma are rare lesions; therefore, this may not be a coincidental presentation. It is thought to have occurred in the embryonic period presumably between 37 and 40 gestational days.

Adult↗
PubMed1994

Hypothalamic hamartomas: with special reference to gelastic epilepsy and surgery.

This study presents six patients with hypothalamic hamartomas diagnosed on the basis of magnetic resonance imaging. Histological confirmation was performed in three patients who underwent surgery. Immunohistological assays were used to determine the neurosecretory pattern. Four patients presented with epilepsy, including gelastic seizures. Other symptoms included behavioral abnormalities in four patients and precocious puberty and visual impairment in two patients. One patient presented associated developmental defects. Good results without morbidity were achieved with surgical resectioning in two patients with large hamartomas associated with behavioral abnormalities and gelastic epilepsy that was unresponsive to conventional medical treatment and in one patient with visual impairment. We propose a classification of the hypothalamic hamartomas based on topographical and clinical data obtained from 36 selected cases in the literature and six of our own cases. This classification should help to classify the various treatment methods and the surgical risks into four subgroups (Types la, lb, lla, and llb). We conclude that the surgical approach is a realistic alternative in certain cases, including large and broad-based Type llb hamartomas associated with gelastic epilepsy and behavioral disorders.

Adult↗
PubMed2006

Hypothalamic hamartoma associated with anterior paraclinoid aneurysm of the internal carotid artery.

A 15-year-old boy presented with a history of medically refractory gelastic seizures and cognitive impairments. Neuroimaging demonstrated a sessile type hypothalamic hamartoma, which was treated by gamma knife surgery. However, the gelastic seizures only partially decreased and the frequency of seizures remained unchanged. One year later, angiography before surgery detected anterior paraclinoid aneurysm of the left internal carotid artery. Blood pressure and endocrinological examinations showed no abnormality. Direct surgery was performed to treat the aneurysm and hamartoma. No sclerotic changes were noted in the arterial wall. The aneurysm was treated with clipping, and the hamartoma was partially removed. Postoperative course was uneventful and the gelastic seizures disappeared. No evidence for a causal relationship between the hamartoma and aneurysm was found.

Adolescent↗
PubMed2002

Surgical treatment of an intramedullary spinal cord hamartoma in a dog.

A 9-year-old spayed female Golden Retriever was examined because of progressive hind limb lameness. Magnetic resonance imaging of the thoracic and lumbar portions of the vertebral column revealed a focal, contrast-enhancing, intramedullary spinal cord mass. The history, signalment, and magnetic resonance findings were suggestive of spinal cord neoplasia. A hemilaminectomy, durotomy, and longitudinal myelotomy were performed, and a 1 X 1-cm mass that contained numerous blood vessels was removed with blunt dissection. Results of histologic examination and immunohistochemical staining of the mass suggested that it was a hamartoma. The dog improved after surgery, with no evidence of a recurrence of clinical signs 14 months after surgery. Vascular malformations of the CNS in dogs include hamartomas, hemangiomas, angiomas, hemangioblastomas, meningocerebral hemangiomatosis, and arteriovenous malformations. A hamartoma is a non-neoplastic overgrowth of cells or an improper proportion of cells that are normally in the involved tissue. Although magnetic resonance imaging may be helpful in determining the extent of the lesion in dogs with vascular malforrmations, it cannot be used to distinguish neoplastic from non-neoplastic formations. Excision may result in a good outcome for dogs with an intramedullary spinal cord hamartoma.

Animals↗
PubMed2006

Spontaneous rupture of splenic hamartoma in a patient with hepatitis C virus-related cirrhosis and portal hypertension: a case report and review of the literature.

Spontaneous rupture is a rare complication of splenic hamartoma. A review of the literature revealed only four such cases. To the best of our knowledge, this is the first report of spontaneous rupture of splenic hamartoma associated with liver cirrhosis and portal hypertension. A 53-year-old woman, who was followed up for aortic dissection and hepatitis C virus (HCV)-related liver cirrhosis, was referred with sudden left chest and shoulder pain. An abdominal ultrasound showed intraabdominal bleeding, and computed tomography indicated rupture of a splenic tumor. Emergent splenectomy was carried out. The postoperative course was uneventful, and the patient was discharged on the 13th postoperative day. Pathology revealed the tumor to be a ruptured splenic hamartoma. The non-tumorous splenic parenchyma revealed congestive changes. We consider that the presence of liver cirrhosis and portal hypertension are risk factors for spontaneous rupture of the splenic hamartoma.

Female↗
PubMed2006

Bile duct hamartomas (von Mayenburg complexes) mimicking liver metastases from bile duct cancer: MRC findings.

We present a case of a 72-year-old man with a common bile duct cancer, who was initially believed to have multiple liver metastases based on computed tomography findings, and in whom magnetic resonance cholangiography (MRC) revealed a diagnosis of bile duct hamartomas. At exploration for pancreaticoduodenectomy, liver palpation revealed disseminated nodules at the surface of the liver. These nodules showed gray-white nodular lesions of about 0.5 cm in diameter scattered on the surface of both liver lobes, which were looked like multiple liver metastases from bile duct cancer. Frozen section of the liver biopsy disclosed multiple bile ducts with slightly dilated lumens embedded in the collagenous stroma characteristics of multiple bile duct hamartomas (BDHs). Only two reports have described the MRC features of bile duct hamartomas. Of all imaging procedures, MRC provides the most relevant features for the imaging diagnosis of bile duct hamartomas.

Aged↗
PubMed1990

Sonographic features of breast hamartomas.

The sonographic features of ten breast hamartomas are reported. Four masses were clinically palpable. The mammographic appearance was characteristic of hamartomas in five cases. In the remaining five lesions, because the radiographic appearance was not pathognomonic, surgical excision was required for pathologic confirmation of the diagnosis. A wide spectrum of sonographic appearances was identified, the most frequent was that of a moderate to well-circumscribed, solid, hypoechoic mass with posterior acoustic shadowing. Two isoechoic hamartomas were very difficult to visualize on sonography. We conclude that ultrasound has a minimal role in the diagnosis of breast hamartomas, in view of their wide sonographic variability.

Adipose Tissue↗
PubMed1999

Congenital non-familial unilateral basaloid follicular hamartoma.

Basaloid follicular hamartoma is not a well-recognized clinical entity and has often been diagnosed as trichoepithelioma or basal cell carcinoma. It is a unique benign follicular tumour which comprises a variety of clinical manifestations. We present the case of a 24-year-old male with unilateral basaloid follicular hamartoma present at birth and later misdiagnosed as basal cell carcinoma. Histological features of basaloid follicular hamartoma are not always diagnostic and clinico-pathological correlation is particularly important to distinguish this benign hamartoma from other basaloid tumours including basal cell carcinoma. Continuous follow-up of our patient did not reveal any clinical or histological malignant transformation.

Adult↗
PubMed1999

[Fibrous splenic hamartoma: imaging features].

Splenoma or splenic hamartoma is a rare primary splenic tumor most often incidentally discovered. The authors report the case of a splenic hamartoma, developed in an asymptomatic man and imaged by US, CT and MRI. This tumor which measured 7 cm in diameter, was hypoechoic with posterior hardening of the ultrasound beam, and not much vascularized as demonstrated by Duplex and color Doppler examination. The tumor MR imaging showed hyperintensity on T1-weighted and hypointensity on T2-weighted. The final diagnosis was not established until the histologic examination of the splenectomy specimen was performed. Histopathologically, the tumor corresponded to a fibrous type of splenic hamartoma. The atypical imaging features which led to splenectomy were probably related to the fibrous component of the hamartoma.

Adult↗
PubMed1997

Muscular hamartoma of the breast: a rare breast lesion containing smooth muscle.

Hamartoma of the breast is an uncommon entity, usually presenting as a well-demarcated breast mass. Microscopically, the lesion is composed of mammary glandular component, fibrous stroma, adipose tissue, and smooth muscle in variable proportions. Among the variants of breast hamartoma, muscular hamartoma is rare. This lesion should be differentiated from other breast tumors that contain smooth muscle element. We report a breast lesion of a 36-year-old woman diagnosed as a muscular hamartoma in which the muscular component is cellular and some mitotic figures are present. The criteria to distinguish between benign and malignant smooth muscle lesions in the breast, emphasizing mitotic count, are also discussed.

Adult↗
PubMed2001

Thrombocytopenia caused by splenic hamartoma: resolution after splenectomy.

We report a rare case of isolated thrombocytopenia due to a large hamartoma of the pulposal type, which resolved with splenectomy in a 46-year-old man. Although hamartomas are usually found incidentally, they may cause hematologic disturbances such as thrombocytopenia, anemia, or splenic rupture. The diagnosis of splenic hamartoma is difficult to make preoperatively. Splenectomy is important in excluding malignant tumors from the diagnosis when the etiology of a splenic mass is unclear and ameliorating hematologic symptoms related to hamartomas.

Diagnosis, Differential↗