Skull fracture mimicking eosinophilic granuloma.

BACKGROUND: Delayed swelling after skull fractures is an uncommon complication following head trauma in children. Classically, growing skull fractures typically present in patients under 3 years of age with progressive subcutaneous fluid collections, or occasionally with neurologic symptoms. We present the case of a healthy 2-year-old boy with a lytic "punched-out" frontal skull lesion. The child presented 2 months after a minor forehead injury for which no medical attention was sought. METHODS: The skull defect had no associated leptomeningeal cyst or brain herniation. Imaging and presentation were thought to be consistent with eosinophilic granuloma. Histologic findings demonstrated a healing skull fracture. RESULTS: Cranioplasty was performed, and the patient had an uncomplicated postoperative course. CONCLUSIONS: In this report, we describe our experience with this atypical presentation of a healing skull fracture mimicking a typical eosinophilic granuloma.

Traumatic ulcerative granuloma with stromal eosinophilia around mini dental implants without the protection of a denture base.

This is a report of a case of an unusual oral lesion after the placement of mini implants for the retention of a mandibular overdenture. A patient received four 2-mm-wide dental implants in the anterior mandible and had her mandibular denture relined with a soft material. After 3 months, she was not wearing her mandibular denture, and two nodular ulcerated lesions were observed near the mini implants. The lesions ceased following excision and regular denture wearing. Clinical and microscopic examination led to the diagnosis of traumatic ulcerative granuloma with stromal eosinophilia (TUGSE). TUGSE is rare lesion with a benign course that may occur following injury of the oral mucosa by mini implants under certain circumstances.

Eosinophilic granuloma of spine in adults: a report of 30 cases and outcome.

BACKGROUND: Eosinophilic granuloma (EG) of the spine is rare, especially in adults. There had been few large and long-term studies reported in the literature. The management goals of this disease in adults are preservation of neurologic function, relief of pain and reconstruction of spinal stability. However, there are still controversies over appropriate management modality of eosinophilic granuloma. METHODS: Clinical manifestations, radiographic presentations, therapeutic outcomes and follow-up findings of 30 adults who were histiologically diagnosed with spinal eosinophilic granuloma, including 28 patients who received surgical treatment at our institutions from 1985 to 2008 were reviewed retrospectively. RESULTS: There were 25 males and five females with a mean age of 34.5 years (range, 18-71 years). The post-operative follow-up period ranged from 2 to 22.4 years (mean, 8.3 years). Neurologic deficits developed in 21 patients, apparent kyphosis developed in four cases. In contrast to the classic feature of vertebra plana in children, we found that more severe lesions often led to asymmetric collapse in adult patients and only three patients presented with vertebra plana. Thirty-three vertebral lesions distributed throughout the spine column. Twenty-one lesions were in cervical spine, seven in the thoracic spine and five in the lumbar spine. Twenty-eight adult patients underwent surgical resection with or without chemotherapy or radiotherapy, and four (13.3%) patients had recurrence after surgery. No patient in our series died. CONCLUSIONS: The onset of spinal EG is insidious and mainly presents as osteolytic destruction. There is a particular high prevalence of lesions in the cervical spine and more severe lesions often led to asymmetric collapse. As the skeleton of adults is well-developed and the epiphysis has stopped growing, individualized management including surgical intervention should be considered in adult patients with spinal EG who present with neurological damage and spinal instability.

Eosinophilic angiocentric fibrosis of the orbit.

A 30-year-old woman complained of right-sided epiphora for 2 years. She also reported diplopia on certain gaze positions and felt a hard mass behind the right medial inferior orbital rim. Magnetic resonance imaging studies demonstrated a relatively well-delineated mass in the right inferomedial orbit with minimal ethmoid sinus involvement. Histopathological evaluation following a large incisional biopsy showed massive eosinophilic infiltration and fibrosis with the final diagnosis of eosinophilic angiocentric fibrosis. She was then discovered to have significant peripheral eosinophilia and elevated serum IgE levels and clinical findings of allergic rhinitis and sinusitis. She was treated with systemic fluorocortolon and desloratadin for 4 months. She remained stable without recurrence for 32 months. The patient with this exceptionally rare tumour of the orbit benefited from debulking surgery followed by systemic corticosteroids and antihistaminics.

Increased pulmonary neuroendocrine cells with bombesin-like immunoreactivity in adult patients with eosinophilic granuloma.

Cigarette smoking is associated with hyperplasia of pulmonary neuroendocrine cells and variably increased levels of bombesin-like peptides in the lower respiratory tract. Because the neuropeptide bombesin is a chemoattractant for monocytes and a mitogen for 3T3 fibroblasts, we hypothesized that an excess of neuroendocrine cells and bombesin-like peptides could contribute to lung inflammation and fibrosis in certain cigarette smokers. Eosinophilic granuloma is a fibrotic lung disease of unknown etiology that in adults occurs almost invariably in cigarette smokers. We quantitated neuroendocrine cells with bombesin-like immunoreactivity in open lung biopsies from patients with eosinophilic granuloma (n = 6) and compared these with cigarette smokers (n = 6) who underwent lung resection for reasons other than primary lung disease. In addition, we compared them with patients with idiopathic pulmonary fibrosis (n = 8), a disease not associated with cigarette smoking. Finally, we also examined the mitogenic effect of bombesin on cultured human adult lung fibroblasts. The patients with eosinophilic granuloma exhibited a 10-fold increase in neuroendocrine cells with bombesin-like immunoreactivity compared to both smokers (P = 0.005) and patients with idiopathic pulmonary fibrosis (P = 0.005). In addition, bombesin produced a significant mitogenic effect on cultured human adult lung fibroblasts at concentrations of 1 nM and above. We conclude that increased numbers of pulmonary neuroendocrine cells with bombesin-like immunoreactivity are commonly found in patients with eosinophilic granuloma and, since bombesin-like peptides are chemotactic for monocytes and mitogenic for human lung fibroblasts, we speculate that neuroendocrine cell hyperplasia may be important in the pathogenesis of eosinophilic granuloma in adult cigarette smokers.

Diffusing capacities and ventilation: perfusion ratios in patients with the clinical syndrome of alveolar capillary block.

Studies were performed on 10 patients with the clinical syndrome of alveolar capillary block while each patient was breathing four different inspired oxygen mixtures. The data were interpreted using the principle of the Bohr integral isopleth with which alveolar oxygen tension in the differently ventilated parts of the lung can initially be treated as unknown. It is then possible to determine the distribution of ventilation, of perfusion, of diffusing capacity, of lung volume, and of alveolar and end capillary blood oxygen tension in the variously functioning parts of the lung. In two patients shunts were the major factor interfering with oxygen transfer. In four others inequalities in ventilation: perfusion ratios and in diffusing capacity in different parts of the lung were the factors interfering with oxygen transfer. In four more patients ventilation: perfusion ratios were the same throughout the lung, the only disturbance of oxygen transfer being in the total diffusing capacity or in its distribution between the different parts of the lung.

Solitary spinal eosinophilic granuloma in children.

Solitary vertebral eosinophilic granuloma is said to be a rare spinal condition in children. 'Vertebra plana' is considered as the typical radiological feature of the involved vertebra; but it could be observed only in 40% of the patients. We have studied three pediatric patients with solitary vertebral eosinophilic granuloma. Their complaint was pain and/or stiffness of the back. Two of them developed neurological deficits in their lower limbs. The diagnosis in imaging (plain radiographs, computed tomography scan and magnetic resonance imaging) varied from one case to another. Histopathology confirmed the diagnosis of eosinophilic granuloma. These three patients underwent an open biopsy and a resection of the spinal lesion achieved with or without interfixation. The outcome was satisfactory in these three cases after a mean follow-up period of over 2 years (from 23 to 27 months).

The prognosis of polyostotic eosinophilic granuloma.

Nine examples of polyostotic histiocytosis without visceral involvement with long-term follow-up are presented. Only one patient had the complete Hand-Schüller-Christian disease triad of multiple osteolytic lesions, diabetes insipidus, and exophthalmos. Three of the nine patients had solitary osseous lesions when first seen but other lesions developed within ten years. The other six had multiple lesions when first seen. In no case was progression to systemic histiocytosis observed. Eight patients were well and in good health seven to 18 years after initial diagnosis. One died indirectly of his disease. Low-dose radiation therapy seems to accelerate healing of lesions.

Eosinophil chemotactic factor in schistosome eggs: a comparative study of eosinophil chemotactic factors in the eggs of Schistosoma japonicum and S. mansoni in vitro.

Significant chemotactic activity for eosinophils was detected in soluble egg antigen (SEA) preparations of both Schistosoma japonicum and S. mansoni in dose-dependent fashion. The activity of S. japonicum SEA was higher than that of S. mansoni SEA. Gel filtration on Sephadex G-150 showed that S. japonicum SEA was composed of two groups of eosinophil chemotactic factors (ECFs), one of high molecular weight (JEE-H) and the other of low molecular weight (JEE-L). S. mansoni SEA showed ECF composition similar to that of S. japonicum SEA. JEE-H was stable on heating (100 degrees C, 60 min) and resistant to pronase digestion, but was sensitive to periodate oxidation. JEE-L was also stable on heating and resistant to pronase and carboxypeptidase A digestions. These properties of the ECFs were also held in common with those of S. mansoni SEA. JEE-L was extractable by toluene, indicating a hydrophobic nature. These results suggest that schistosome eggs themselves contain ECFs, and that the composition of S. mansoni and S. japonicum SEA-derived ECFs is essentially the same. However, they differ from the other ECFs which have already been described in schistosome infections.

Schistosoma mansoni infection causing diffuse enteric inflammation and damage of the enteric nervous system in the mouse small intestine.

Schistosomiasis mansoni is a major health problem, mainly occurring in developing countries. A large proportion of infected individuals suffers from motility-related gastrointestinal problems. In the present study, the diffuse inflammatory response in the small bowel wall, as compared to the egg-induced granulomatous inflammation, was investigated. For this purpose, OF1 mice infected with Schistosoma mansoni 8-16 weeks prior to the experiment, and uninfected control mice were studied. The ileum showed both a diffuse mucosal inflammation as well as a granulomatous reaction. The diffuse mucosal inflammation caused an increase in the thickness of the mucosa, with blunting of the villi. A significant, transient increase of thickness of the muscularis propria after 12 weeks of infection was noted. There was an infection-related mast cell infiltrate in the muscularis propria, consisting of formalin fixation-insensitive connective tissue mast cells. Ganglionitis of the myenteric plexus was noted. Rarely, ganglia of the myenteric plexus contained apoptotic cells. A general pharmacological set of experiments showed a significant increase in intestinal contractility, both to exogenously administered, as well as to endogenously released neurotransmitters. Our results demonstrate that S. mansoni infection in the mouse ileum leads to diffuse specific enteric inflammation that is associated with an enhanced response to contractile agents.

Other smoking-affected pulmonary diseases.

Cigarette smoking is the leading cause of preventable death in the United States. Smoking adversely affects many organ systems, but especially the lung. Carcinoma of the lung and chronic obstructive pulmonary disease account for most smoking-associated respiratory morbidity and mortality, and their association with smoking is both well established and widely recognized. Cigarette smoking also is associated with differences in the incidence, severity, or natural history of a broad array of other respiratory illnesses, ranging from the common cold to pneumothorax, pulmonary hemorrhage, and various interstitial lung diseases. Interestingly, while the general effect of smoking on respiratory diseases is adverse, in the cases of sarcoidosis and hypersensitivity pneumonitis smoking may actually be associated with a decrease in the incidence of disease. In this article, the author briefly discusses some of the pulmonary and systemic effects of smoking that might mediate its effects on an array of lung diseases, then comprehensively reviews less common or less well-recognized smoking-affected lung diseases such as pulmonary infections, spontaneous pneumothorax, Goodpasture's syndrome, eosinophilic granuloma and other interstitial lung diseases, and pulmonary metastatic disease.

Eosinophilic granuloma. A different behaviour in children than in adults.

Localised Langerhans-cell histiocytosis of bone (eosinophilic granuloma) is a benign tumour-like condition with a variable clinical course. Different forms of treatment have been reported to give satisfactory results. However, previous series all contain patients with a wide age range. Our aim was to investigate the effect of skeletal maturity on the rate of recurrence of isolated eosinophilic granuloma of bone excluding those arising in the spine. We followed up 32 patients with an isolated eosinophilic granuloma for a mean of five years; 17 were skeletally immature. No recurrences were noted in the skeletally immature group even after biopsy alone. By contrast, four of 13 skeletally mature patients had a recurrence and required further surgery. This suggests that eosinophilic granuloma has a low rate of recurrence in skeletally immature patients.

Eosinophilic granuloma associated with intratumoral hemorrhage--case report.

A 2-year-old boy presented with a rapidly growing soft scalp mass in the left parietal region. Surgical exploration and histological examination demonstrated an eosinophilic granuloma associated with intratumoral hemorrhage. Eosinophilic granuloma should also be considered when a scalp mass lesion exhibits rapid growth.

[Rapid remission of pulmonary eosinophilic granuloma in a young male patient after cessation of smoking].

A 24-year-old asymptomatic man was referred to our hospital in October 7, 1997 for further examination of abnormal shadows detected on chest X-ray films that had been obtained during an annual health examination on September 12, 1997. The patient had suffered productive cough since the end of August 1997. A chest X-ray film obtained one year earlier showed no abnormalities. The patient had smoked 20 cigarettes per day since the age of 14. The findings of a physical examination and laboratory tests were unremarkable. A chest X-ray films showed micronodules 2-5 mm in size that were predominantly distributed in the upper and middle fields of both lungs. A chest computed tomogram (CT) showed multiple cysts measuring about 10 mm in diameter, with thick walls (2 to 3 mm) in addition to small nodular shadows. A transbronchial lung biopsy and a thoracoscopic lung biopsy revealed several stellate nodular fibrotic lesions containing some S-100 protein-positive, large mononuclear cells and lymphoid cells. An electron microscopic study found several Langerhans' cells with Birbeck's granules in their cytoplasm. A chest CT scan obtained 2 months after the patient stopped smoking (the day of admission) showed marked regression of the cystic shadows. Evidence suggests the pathogenesis of the disease is closely linked to smoking, and some case reports have documented clinical and radiographic improvements after patients stop smoking. Pulmonary small nodular lesions and cystic lesions with thick walls tend to undergo regression. Although surgical lung biopsy specimens disclosed several stellate nodular fibrotic lesions of eosinophilic granuloma that seemed to be histologically irreversible, our patient experienced remarkable regression of his pulmonary lesions within 2 months after the cessation of smoking.

[Clinical course of pulmonary eosinophilic granuloma--CT evidence of lesion evolution].

We reviewed a clinicopathological study of pulmonary eosinophilic granuloma focusing on the evolution of pulmonary lesions in chest CT scans. Between 1990 and 2002, five patients with pulmonary eosinophilic granuloma were admitted to our hospital. Clinical features, chest radiographs, pathological characteristics and treatments were evaluated. The scans were repeated in four patients at intervals of at least eight months. All patients were men who smoked. The age at onset of the pulmonary disorders ranged from 22 to 45 years, with a mean of 33 years. Most of the initial chest CT scans showed nodular lesions and thin-walled cysts, but the nodular lesions disappeared or were transformed into cystic lesions in the later scans. Most of the final scans revealed thin-walled cysts and emphysematous lesions. The histopathological characteristics of the nodular lesions in the chest CT were correlated with cellular granulomas mainly composed of Langerhans cells and eosinophils, and small granulomas were also seen in the thin fibrous walls of the cysts. All patients quitted smoking and three showed improvement of the pulmonary lesions within three months.

[Oral histiocytosis X with severe upper maxillary destruction]. / Histiocitosis X oral con severa destrucción del maxilar superior.

Histiocytosis X is a group of disorders of the reticuloendothelial system that have different clinical features and common histological characteristics, including abnormal proliferation of Langerhans cells. Eosinophilic granuloma is a localized form of histiocytosis affecting one or more bones. A case report is made of a patient diagnosed as histiocytosis X whose first manifestation was the appearance of an orosinusal communication. The lesions were located in the maxillary bones and produced large osteolytic lesions. No other bones were affected. In spite of surgery and radiotherapy, the disease recurred at 12 months.