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Biomedical subjects

M Caldarelli

Publications and source records attributed to M Caldarelli.

At least 19 recordsLinked to original sources

Bone mineral density in survivors of childhood brain tumours.

BACKGROUND: Osteopenia has been reported in children surviving acute lymphoblastic leukaemia, apparently as consequence of therapy. Few studies have been published on bone mineral density (BMD) evaluation in children surviving from brain tumours. The endocrine system in these patients is frequently affected as consequence of therapeutic interventions such as cranial irradiation and anti-neoplastic agents: growth hormone deficiency is the most common adverse sequel. The pathogenesis of osteopenia in brain cancer survivors is multi-factorial but still uncertain. OBJECTIVE: The aim of this study is to examine bone mass in 12 brain cancer survivors and its relationship with their hormonal status. RESULTS AND DISCUSSION: We observed that most of the patients had a BMD that was lower than normal in both the lumbar column and in the femoral neck. Bone mass loss was higher in the lumbar region rather than in the femoral neck, due to spinal radiation therapy and to the effect of hormonal deficiencies. Particularly hypogonadism, but also multiple hormonal deficiencies, are associated with lower BMD values. Experience in clinical care of these patients suggests the importance of periodic evaluations of BMD, especially in those with secondary hormone deficiencies. Moreover, the periodic assessment of the hypothalamus-pituitary function is essential for an early diagnosis of hormonal insufficiency, primarily hypogonadism, to precociously detect bone mineral loss and to prevent pathological fractures, thus improving the quality of life.

Adolescent↗

Hydrocephalus in CINCA syndrome treated with anakinra.

INTRODUCTION: Chronic infantile neurologic, cutaneous, articular (CINCA) syndrome is a rare congenital autoinflammatory disease characterized by neonatal-onset chronic meningitis, hydrocephalus, sensorineural hearing loss, persistent urticarial rash, deforming arthritis, and recurrent fever. This clinical entity is believed to result from dysregulation of cytokine production. No recommended treatment protocol exists so far for CINCA syndrome. CASE REPORT: We report a 7-year-old child affected with CINCA syndrome in whom no therapy had resulted effective. Anakinra, an interleukin-1-receptor antagonist, was administered in a 1-year period with complete inflammatory symptom remission and dramatically ameliorated laboratory tests. This optimal response has been supported by the demonstration of a stabilized hydrocephalus upon magnetic resonance imaging and by an overall improvement of the neurodevelopmental issues. DISCUSSION: This paper emphasizes and discusses the medical approach with anakinra in CINCA syndrome presenting with hydrocephalus in which a consistent control of the neurological picture can be obtained.

Arthritis↗

Phase II trial of temozolomide in children with recurrent high-grade glioma.

PURPOSE: The objective of the study was to evaluate the efficacy and toxicity of Temozolomide (TMZ) administered for 5 consecutive days in three daily dosing in children with recurrent or refractory high-grade glioma. PATIENTS AND METHODS: Twenty-four patients with a median age of 10.5 years were enrolled onto this open-label, multicenter, phase II study. The patients were previously treated with surgical resection (17 of 24), radiotherapy (19 of 24) and chemotherapy (18 of 24). Therapy was administered orally three times a day for 5 consecutive days at the dose of 200 mg/m(2)/dx5 for chemotherapy naive patients. In patients heavily pretreated with chemotherapy the starting dose was of 150 mg/m(2)/dx5. RESULTS: A total of 95 cycles were administered. The median progression free-survival (PFS) was 3 months for the entire group while disease stabilization was obtained in 7 patients (29.1%), all with supratentorial tumors. No CR or PR was observed. TMZ treatment showed a limited toxicity. Thrombocytopenia was the most common hematological adverse effect. Our data suggest a marginal activity of TMZ in children with recurrent high-grade glioma.

Adolescent↗

Long-term results of the surgical treatment of craniopharyngioma: the experience at the Policlinico Gemelli, Catholic University, Rome.

BACKGROUND: Craniopharyngioma (CP) is the most common intracranial non-glial tumour observed in pediatric age. Although histologically benign and amenable to surgical treatment, its location and relation with vital nervous and vascular structures makes the feasibility of a radical resection difficult even in the microneurosurgery era. Beside the difficulties experienced when performing tumour resection, post-operative complications, such as endocrinological imbalance, represent another point that makes CP total excision a challenge. In order to avoid such complications, some authors have suggested to renounce to radical resection and to rely on post-operative radiation therapy to minimise the risk of residual tumour progression. METHODS: We report our experience with 52 children and adolescents operated on for CP at the Department of Pediatrics, Section of Pediatric Neurosurgery, Catholic University Medical School, Rome, between January 1985 and December 2002. The study included 14 children <5 years old (five less than 2 years of age), 25 between 6 and 10 years old, and 13 more than 10 years old. The most common presenting signs were related to endocrinological imbalance (35 cases), increased intracranial pressure (32 cases), and to a lesser extent, visual compromise (17 cases). Concerning location, CP was intrasellar in three cases; sellar/suprasellar with prominent prechiasmatic growth in 24 cases; retrochiasmatic/3rd ventricular in 14 cases, and giant (with an extension into the middle and/or posterior cranial fossae) in 11 cases. The tumour was managed by means of a single surgical approach in 47 cases and with a two-stage operation in the remaining five cases. In 11 cases of intrasellar or intra/suprasellar midline location, the first surgical approach was done through the transsphenoidal route (which represented the first step of a staged operation in five instances); in the remaining 41 patients, craniotomy was the first surgical procedure. Radical tumour resection was achieved in 40 cases, subtotal (only small tumour remnants adherent to the carotid arteries, 3rd ventricle floor or visual pathways) in nine, and only partial in the remaining three cases RESULTS: Histology demonstrated the adamantinous variant in all cases. Two surgical deaths were recorded in this series (both following a transsphenoidal approach): one secondary to uncontrollable intra-operative bleeding from the carotid artery, and the other to fulminating bacterial meningoencephalitis. Morbidity included endocrinological disturbances, namely hypopituitarism and diabetes insipidus, in more than 80% of cases, worsening of pre-operative visual deficit in six cases, and transitory neurological deficits in five cases. One late death was recorded 2 years after surgery, secondary to electrolytic imbalance although favoured by a major head trauma with subacute subdural haematoma. Nine recurrences occurred 1-8 years after surgery (three true recurrences, and six re-growths of incompletely resected tumours) that required re-operation. Twelve patients underwent radiotherapy, six after an initially incomplete tumour resection and six following relapse. One patient presented with a malignant thalamic glioma 8 years after radiotherapy. At long-term follow-up, all survivors showed good clinical condition, even though approximately 60% relied on hormone replacement, and some patients presented obesity. Diabetes insipidus has subsided in about 80% of the cases; visual deficits improved or remained stable, whereas post-operative neurological deficits subsided in all but one patient. CONCLUSION: In our experience, radical resection of CP represented the first and almost unique treatment modality. Although not insignificant, post-operative mortality and morbidity do not seem to represent a major contraindication in attempting a radical tumour resection whenever possible. On the other hand, extensive hypothalamic involvement should suggest a less aggressive attitude.

Adolescent↗

Intracranial pressure monitoring in children with single suture and complex craniosynostosis: a review.

INTRODUCTION: One third of patients with craniofacial dysostosis syndromes and about 15-20% of children with single suture craniostenosis have a documented increase in intracranial pressure (ICP). The early detection of intracranial hypertension is important in order to reduce the risks for brain development and visual function. However, in children with craniosynostosis, the clinical manifestations of abnormally increased ICP are difficult to detect, as the majority of patients may have neither warning signs nor symptoms for a long period of time. REVIEW: Moreover, data from the literature suggest that neither fundoscopic nor radiological findings are necessarily related to intracranial pressure recordings in this type of pathology. In this context prolonged ICP monitoring seems actually to be the most valuable diagnostic tool. Extradural, subdural, and intraparenchymal devices have been used, and different softwares have been employed for PC storage and analysis of the data obtained. Most recent series consider mean ICP combined with plateaux waves as the most sensitive indicators in the final evaluation of the results. CONCLUSIONS: The analysis of the literature demonstrates a significant difference in the prevalence of raised ICP preoperatively between non-syndromic and syndromic patients. Among the non-syndromic children, a direct relation between the number of sutures involved and raised ICP is documented. The analysis of plateaux waves seems to be particularly important in children with borderline mean ICP values. Persistent postoperative raised ICP has been described in 6-15% of patients with craniofacial dysostosis. It must be related to the multifactorial etiology of increased ICP in these patients, which includes cerebral venous congestion, upper airway obstruction, and hydrocephalus.

Cerebrospinal Fluid Shunts↗

Fibromyxoid sarcoma in a 4-year-old boy: case report and review of the literature.

The authors report on the case of a low-grade fibromyxoid sarcoma (LGFMS) with giant rosettes in a 4-year-old boy. The tumor arose in the paravertebral region and had infiltrated the vertebral canal between L2 and S1. A review of the literature indicates that this is one of the youngest patients diagnosed with a tumor of this type, and spinal involvement had never been reported with LGFMS.

Child, Preschool↗

Latex sensitisation and allergy in children with myelomeningocele.

INTRODUCTION: Children with spina bifida (SB) have a high degree of exposure to latex products as a consequence of repeated surgical procedures, implantation of latex-containing materials and catheterisation. The consequence is a higher incidence of latex allergic reactions. OBJECTIVE: The aim of this study is to evaluate the prevalence of latex sensitisation and allergy in a population of children with myelomeningocele (MMC) and to assess the role of associated risk factors. RESULTS: Forty-eight percent of the patients (29 out of 60) showed a latex sensitisation with specific IgE >0.7 kU/l while 15% (9 out of 60) were allergic to latex (specific IgE >0.7 kU/l and clinical manifestations). The principal factor correlated with allergy to latex was specific serum IgE to latex (radioallergosorbent test [RAST]) values (p<0.01). Other factors were total serum IgE (paper radioimmunosorbent test [PRIST]) values, number of surgical procedures and familiarity with allergy. CONCLUSION: These results underline the importance of prophylactic measures to avoid the exposure, not only in the sanitary environment, through the institution of latex-safe routes, but also in daily life, to prevent potentially serious allergic reactions.

Adolescent↗

Perioperative management of face-to-face craniopagus twins separation.

Craniopagus conjoining represents a complex and challenging issue for neurosurgeons as well as for anesthesiologists. A rare face-to-face case of conjoined twins underwent surgical separation and presented peculiar differences compared with those already reported in the literature. Even in cases lacking large cerebrovascular sinus connections, the impending risk of large blood loss and hemorrhagic shock in the infant requires a high level of surveillance and the institution of invasive monitoring.

Anesthesia, Inhalation↗

Craniopagus: the Thessaloniki-Rome experience.

OBJECT: A case of two female Greek twins conjoined at the left frontal and orbital regions is described. These conjoined twins were transferred to the Pediatric Neurosurgical Unit of the Gemelli Hospital, Catholic University Medical School, at the age of 4 months for possible surgical division. METHOD: Multiple diagnostic investigations, which included in particular advanced imaging techniques, demonstrated an independent cerebral vascularization of the two babies and excluded significant intracranial malformations. Such an accurate evaluation of the anatomy of the craniopagus at the level of the cranial junction allowed the feasibility of the surgical correction to be established. CONCLUSIONS: The operation was carried out by a multidisciplinary team in a single stage. The operating team included two distinct teams of neurosurgeons and anesthesiologists as well as a plastic surgeons' team. Both babies survived the operation. The postoperative course was uneventful. Both twins were discharged in good clinical and neurological condition after a 3-week period of hospitalization.

Brain↗

Risk factors for atherogenesis in children with spina bifida.

INTRODUCTION: Several literature reports have demonstrated that cardiovascular disease is one of the most frequent pathologies in patients with spinal cord injury. AIM: The aim of this study is to reveal changes in the lipid profile in non-obese children with spina bifida/myelomeningocele (MMC). DISCUSSION: The original results demonstrate the necessity of metabolic surveillance in these patients. Daily physical activity must be ensured early on frequency in non-able-bodied children in order to prevent changes in seric lipoproteins.

Adolescent↗

Muenke syndrome.

BACKGROUND: Muenke syndrome is a genetically determined craniosynostosis that involves one or both coronal sutures. In some patients it is associated with skeletal abnormalities such as thimble-like middle phalanges, coned epiphysis, and/or neurological impairment, namely sensorineural hearing loss or mental retardation. In spite of a variable phenotype, Muenke syndrome has been related to a unique mutation on the FGFR3 gene, Pro 250 to Arg, which is characteristic of this disease. Because of the incomplete penetrance of this anomaly, the suspicion of Muenke syndrome must be raised in any child with uni- or bilateral coronal craniosynostosis, and the genetic analysis propounded even in the absence of extracranial features. ILLUSTRATIVE CASES: We report the cases of two sisters who presented with Muenke syndrome and whose affected mother did not have any form of craniosynostosis.

Arginine↗

[Endoscopic third ventriculostomy: complications and failures].

Endoscopic third-ventriculostomy (ETV) became the treatment of choice for non-communicating hydrocephalus and its effectiveness is largely reported. On the contrary, specific articles on complications and failures of this technique are very rare and this review aims at supplying further information about it. Therefore, an analysis of the main and up-to-date series is made and exhaustive data about complications and failures of ETV and about their incidence are obtained. The overall frequency rate of complications is 6-20%. Their severity may vary either because of the length of the damage (transient or permanent) or the value of the involved structures (basilar artery, areas of the CNS, hypothalamus) or the importance of the injury (from subclinical sequelae to fatal complications). Moreover, this study showed both the risk that the endoscopic procedure must be suspended (intra-operative failure) and the risk of an early (before 1 month) or delayed (even some years after the intervention) narrowing of the ventriculostomy. The rate of and the reasons for failure have been analysed and the success of a second ETV has been estimated. The results of this analysis suggest that the children proposed for ETV are carefully selected and meticulously studied during the follow-up.

Blood Loss, Surgical↗

Neurocutaneous melanosis.

BACKGROUND: Neurocutaneous melanosis (NCM) is a rare phakomatosis characterized by a focal or diffuse proliferation of melanin-producing cells in both the skin and the leptomeninges. This syndrome is believed to result from an error in the morphogenesis of embryonal neuroectoderm. FEATURES: Two-thirds of patients with NCM have giant congenital melanocytic nevi, and the remaining third have numerous lesions but no giant lesions. Most patients present neurological manifestations early in life, which can be secondary to intracranial hemorrhages, to impairment of cerebrospinal fluid circulation, and to malignant transformation of the melanocytes. PROGNOSIS: The prognosis of patients with symptomatic neurocutaneous melanosis is extremely poor, even in the absence of malignancy. Chemotherapy has been ineffective in the few patients in whom it has been tried.

Case-Control Studies↗

Subdural hygroma: an unwanted result of Sylvian arachnoid cyst marsupialization.

INTRODUCTION: Subdural hygroma is a known complication of Sylvian fissure arachnoid cysts (SACs). However, most of the data in the literature refer to spontaneous or posttraumatic occurrence of subdural hygromas, regarded as either a favorable or an unfavorable event. Little is known about this phenomenon as a consequence of the surgical management of SACs. The present study was carried out to evaluate the significance of postoperative subdural hygromas in children with temporal arachnoid cysts, who have been treated with craniotomy and wide marsupialization of the cystic membrane. CLINICAL MATERIAL AND RESULTS: Between 1980 and March 2002, 104 children were operated on for a SAC at the Pediatric Neurosurgical Unit of the Catholic University Medical School in Rome. Six patients (5.8%; boys/girls=4/2; mean age 3.28 years) developed a subdural hygroma postoperatively, which required further treatment. According to Galassi's classification 4 children had a Type III cyst and 2 children a Type II cyst. All the patients had previously been submitted to open marsupialization of their arachnoid cyst and extensive removal of the cyst wall. In 5 patients (Type III cyst: 4 patients; Type II cyst: 1 patient), acute or subacute symptoms and signs of increased intracranial pressure (ICP) developed at a temporal distance, varying from 5 days to 1 month (mean interval: 14.3 days). All these children needed surgical treatment for the hygroma. A subduro-peritoneal (SDP) shunt was implanted as the first step in 3 patients (Type III cyst: 2 patients; Type II cyst: 1 patient). In the remaining 2 patients of this group (Type III cysts) medical therapy was initially attempted (acetazolamide: 24 mg/kg) without any improvement in clinical manifestations; on these grounds an external spinal subarachnoid drainage was implanted, but led only to the transient clearance of symptoms. The direct drainage of the subdural collection (SDP shunt in 1 patient and temporary external subdural drainage in the other), was followed by complete clinical recovery in both cases. The last patient in this series showed a pathologic increase in head circumference during the 1st month after surgery for his arachnoid cyst, followed by a subsequent stabilization. A CT scan documented a subdural hygroma, which first of all increased in size, but stabilized 2 months after surgery. No surgical treatment was performed in this case. At a mean follow-up of 2.38 years (minimum: 1 month; maximum: 5 years) all the patients are in excellent clinical conditions. Complete neuroradiological resolution of the hygroma was observed in 2 of the 4 shunted patients, who have both been submitted to shunt removal (2 and 3 years after the implant respectively). CONCLUSIONS: It is our opinion that a wide opening of the external membrane of SACs may predispose the CSF fluid to accumulate within the subdural space, where its absorption is insufficient. A ball mechanism created by CSF pulsation may further increase the subdural fluid accumulation. Secondary distortion and occlusion at the level of the basal cisterns may also contribute to the persistence of the subdural fluid collection. Clinical manifestations may be transient, but frequently have a progressive course and the drainage of the subdural collection is, though transient, required in most cases. On these grounds we suggest the selective opening of the basal cisterns associated with a limited resection of the outer cyst walls in order to limit CSF access to the subdural space.

Arachnoid Cysts↗

Intramedullary epidermoid cyst presenting with abnormal urological manifestations.

STUDY DESIGN: Report of an epidermoid cyst with intramedullary localization. OBJECTIVE: To describe an atypical presentation of intramedullary epidermoid cyst. SUMMARY OF BACKGROUND DATA: Intramedullary epidermoid cysts are rare entities with a marked variability in the clinical presentation, essentially of neurological pertinence. METHODS: Case report of a spinal epidermoid cyst in a 13-year-old girl presenting with urological symptoms: she had a 12-month history of recurrent low urinary tract infections, urinary frequency and nocturnal enuresis. A urodynamic evaluation was performed and showed the presence of involuntary bladder contractions with detrusor instability and low bladder compliance. Magnetic resonance imaging of the spine demonstrated an intramedullary lesion of the dorsal spinal cord. RESULTS: The mass was excised and 6 months after surgical excision, urological manifestations improved with decreased detrusor hyper-reflexia, increased bladder capacity and compliance and no later report of urinary tract infections. CONCLUSIONS: In our patient, unusual clinical manifestations of the tumor have delayed the diagnosis, but its complete removal has led to remission of symptoms. Detailed neurological examination and investigations are indicated in patients with clinical and urodynamic features, suggestive of neuropathic bladder.

Adolescent↗

[Prolonged ICP monitoring in children with sylvian fissure arachnoid cysts].

BACKGROUND: The decision making process for patients with sylvian fissure arachnoid cysts still represents a challenge for the neurosurgeon. A high proportion of the patients is indeed asymptomatic, in spite of neuroimaging signs of apparently increased intracranial pressure (ICP). The aim of the present study was to evaluate the usefulness of prolonged ICP recording in the preoperative work-out. METHODS: Twelve children (11 M/1 F) harboring temporal arachnoid cysts were investigated (men age 6.3 years). According to Galassi classification they were subdivided into 3 groups. Group I included 3 patients with Type I cysts; group II comprised 6 children with Type II cysts; group III consisted of 3 children with Type III cysts. An extensible silicone microprocessor (Codman ), developed for continuous ICP recording, was implanted intraparenchymally, adjacent to the major extension of the cyst. The collected information was cable unloaded to a PC and stored. All the patients underwent a minimum of 48 hours to a maximum of 72 hours ICP continuous check. Ten mmHg was arbitrarily chosen as the upper normal limit of ICP in resting conditions and the patients were classified according to the percentile distribution of their ICP daily and nightly pressure values. RESULTS: ICP recordings were in the normal range in all the 3 children with Type I cysts (49-86% <10 mmHg during the entire recording), in spite of the fact that 2 of them were apparently symptomatic. Three of the 6 children with a Type II temporal cyst had elevated ICP values (69-99% >10 mmHG), even though they had an incidental (1 case) or prenatal (2 cases) diagnosis. The remaining 3 patients had normal ICP values for more than 70% of the recording time period. Two of the 3 patients with type III temporal arachnoid cysts had almost constantly abnormal ICP values (95=99% >10 mmHg); conversely the third patient showed normal ICP values for more than 80% of the recording time period. Five patients were operated on; in 4 of them the surgical indication was based on ICP recordings (2 of the 3 children with a Type II cyst and increased ICP and the 2 patients with a Type III cyst and increased ICP). The last child, harboring a Type I cyst, was operated on under parents request, as a preventive measure. At a mean follow-up of 10.1 months all the patients operated on are in excellent clinical conditions; 1 of them (Type II cyst) initially submitted to craniotomy and cystic membrane excision needed a subdural-peritoneal shunt implant 3 months after surgery for the appearance of a symptomatic subdural hygroma on the side of the cyst. Postoperative CT showed signs of brain expansion in the 2 patients operated on for a Type III cyst, and in the patients operated on for a Type I cyst. CONCLUSIONS: With the limit of the relatively small series here presented, prolonged ICP recording appeared to be an important preoperative tool to rule out the necessity of operating on children with Type I cysts. Furthermore, it was particularly useful in the decision-making process for children with Type III cysts. In cases of Type II lesions, the investigation resulted less discriminating, though the ICP monitoring contributed to find out those patients in whom surgery was indicated in spite of the absence of symptoms.

Adolescent↗