TUGAS BAHASA INGGRIS
MEDICAL CASE REPORT
DISUSUN OLEH:
1. Amilia (P1337430214032)
2. Aisyah Amalia D R (P1337430214061)
PRODI D IV TEKNIK RADIOLOGI SEMARANG
JURUSAN TEKNIK RADIODIAGNOSTIK DAN RADIOTERAPI
POLITEKNIK KESEHATAN KEMENKES SEMARANG
2015
PEDIS RADIOGRAPHIC EXAMINATION IN PATIENT WITH OSTEOARTHRITIS IN HOGWARTS HOSPITAL
INTRODUCTION
Radiographic Examination is examination using X-Ray to provide information about anatomy and phatology. One of radiographic examination that often performed is Pedis radiographic examination. There are three basic projections in pedis; anteroposterior (AP), lateral, axial, oblique, and weight bearing (Bontrager, Fifth Edition 2001)
There are 3 projections used in radiographic examination in Hogwarts Hospital, there are anteroposterior (AP), lateral, and oblique, it can cause inaccurate diagnosis.
The purpose of this case report is to describe Pedis radiographic examination procedure in patient with osteoarthritis in Hogwarts Hospital.
Osteoarthritis (OA) is an abnormality of a joint caused by imbalance physiology which causes damage joints. It's also known as degenerative arthritis or degenerative joint disease or osteoarthrosis, is a group of mechanical abnormalities involving degradation of joints, including articular cartilage and subchondral bone. Osteoarthritis (OA) commonly affects the hands, feet, spine, and the large weight bearing joints, such as the hips and knees, although in theory, any joint in the body can be affected. As OA progresses, the affected joints appear larger, are stiff and painful, and usually feel better with gentle use but worse with excessive or prolonged use, thus distinguishing it from rheumatoid arthritis.
CASE PRESENTATION
Mrs. Ceres (40 years old, 165 cm tall, 50 weight) felt pain since she got high sugar levels in 2010. In her right foot there was a wound in her fingers. That wound was getting worse until now. Injuries incurred after she wearing shoes that were routinely used by her to the workplace. Trauma history denied. Wound pain is felt as throbbing. There was complaint of fever. Every day, she took care of wounds herself at home, and then the wound grew fast and growing pains. She then treated to a clinic, given antibiotics. She diagnosed got osteoarthritis. The doctor suggested her to have radiographic examination first. In this hospital, radiographic examination was done using two projections, AP axial weight bearing and oblique.
Position of patients of AP Axial Weight Bearing Projection was erect, both hands beside the body as comfortable as possible. Flexions legs and soles of the feet facing the desk examination. Put the tape on the floor. The patient stands on the tapes. Position the marker according to the position of the foot. Place a ruler measuring (scale) to make it easier to position the feet so symmetrical. FFD was 90-100cm. CR in 10º (toward os calcaneus), and CP on 3rd of Metatarsal and it's used imaging plate size 24 cm x 30 cm.
Position of patient of oblique projection was supine. Both hands beside the body as comfortable as possible. Give a pillow under the head. Flexions legs and soles of the feet facing the desk examinations. The object was endo rotation foot with 30º angle to the tape on the lateral side. With FFD 90-100cm. CR was 10º (toward os calcaneus) and CP on 3rd of Metatarsal. And imaging plate's size was 24 cm x 30 cm.
DISCUSSION
In literature stated that radiographic examination in Pedis use three projections. But in Hogwarts Hospital only used two projections, AP axial weight bearing and oblique are needed to provide information of base of Pedis that indicate the osteoarthritis.
This radiographic projection is used to see joints space in patient's pedis.
CONCLUSION
Indication of osteoarthritis is the occurrence of aging process of the body organs especially is pedis who was the greatest burden has to sustain weight.
Pedis radiographic examination in patient with osteoarthritis in Hogwarts Hospital was same to the literature, but for osteoarthritis only used two projections, it's AP axial weight bearing and oblique that might cause accurate diagnosis. It's used AP axial weight bearing because this radiographic examination of osteoarthritis in cases of very important for the purpose of comparing the state of both the pedis. The assessment is done by comparing the injured joint space with normal joint space, therefore certain radiographic examination is performed with the patient's position. It's not used lateral projection because this projection not give more benefit for osteoarthritis, it's used to see corpus alienum.
REFERENCES
Bontrager, Kenneth, L. 2003. Text Book Of Radiographic Positioning And Related Anatomy,
Fifth Edition. The Mosby, St. Louis.
http://anjelitahna.blogspot.com/2014/03/bahasa-inggris-radiologi-case-report.html
A PEDIATRIC CASE OF GORHAM'S DISEASE WITH
EXTENSIVE MAXILLOFACIAL INVOLVMENT
ABSTRACT
Objectives: To present a rare pediatric case of Gorham's disease with extensive head and neck involvement.
Study Design: Case report and literature review.
Methods: Literature review of Gorham's disease in pediatric patients with head and
neck manifestations with discussion of a representative case within our health system.
Results: We describe a rare and fatal pediatric case of Gorham's disease originating in the mandible, and progressing over 5 years to involve the maxilla, sphenoid bone, temporal bone, occipital bone, and infratemporal fossa. A nine year old male initially presented after presumed mandibular dental trauma. An insidiously progressive massive maxillofacial osteolysis ensued. The diagnosis of Gorham's disease was established only after infectious, malignant, inflammatory and endocrinologic diseases were excluded. Facial photographs and computerized tomographic images demonstrate the dysmorphic facial features of this vanishing bone disease. The literature is reviewed and medical and surgical management is discussed. Gorham's disease in a pediatric patient with extensive head and neck involvement is exceptionally rare.
Conclusion: To the best of our knowledge our case represents the fourth case in the literature to document Gorham's disease in a pediatric patient with mandibular involvement also photographic images are provided.
INTRODUCTION
Few cases of Gorham disease (GD) have been reported in the literature. It has been called vanishing bone disease, massive osteolysis, phantom bone disease and is also known as Gorham-Stout syndrome. GD was first reported by Jackson in 1838 in his patient description of a boneless arm due to massive osteolysis.14 Romer described the first head and neck case of GD in 1928.26 Less than 150 cases have ever been reported in the literature.15 Its etiology and pathogenesis are unknown. It often presents as a pathologic fracture more commonly in males in their second and third decades of life. No hereditary correlation or racial redilection is known. This disease frequently affects the long bones, pelvic girdle, shoulder girdles, vertebrate, and ribs. Since 1928, forty-two cases involving the maxillofacial region have been documented. Of those, only three have involved the mandible of a male patient younger than ten years of age.
We report the case of a nine year old male suffering from massive osteolysis which after extensive workup was diagnosed as GD. His disease originally manifested in his mandible and progressed to involve his maxilla, sphenoid, temporal bones and infratemporal fossa.
CASE REPORT
RG, is a 9 year old male who suffered mild trauma to the face after falling. He did not sustain any bony fractures. 3 months later, his right mandibular molar teeth were noted to be loose and inflamed requiring extraction. After a biopsy revealed sulfur granules, the patient was treated with periodic debridement and biopsies for presumed Actinomycosis steomyelitis, although no actinomycosis had been cultured.
The patient's mandible underwent continued osteolysis despite aggressive intravenous and oral antibiotics. 18 months later, contralateral right mandibular involvement was noted. 5 months later, the left maxillary molars were found to be loose, and defects in the hard palate as well as nasal speech were present. The mandible showed near-complete osteolysis with only the left ascending ramus intact.
One year later, a magnetic resonance imaging (MRI) scan of the head and skull base
revealed contiguous spread of the osteolysis to include the clivus, petrous mastoid, carotid canal, jugular fossa, right pterygoid plates, sphenoid body and occipital bone with erosion of the floor and anterior wall of the right middle cranial fossa. A computerized tomography (CT) scan the following month showed dehiscence of the right middle cranial fossa floor onto the right infratemporal fossa. Further analysis of the CT scan showed that the patient's right cochlea was at risk of this progressive osteolysis.
Nine months later, this aggressive spread of osteolysis prompted an infraorbital ridge
biopsy, which revealed massive osteolysis without concern for underlying malignancy. His disease progressed without clinical signs of infection, there was no erythema, edema, or leukocytosis. Given the progressive nature of his disease without a identifiable etiology he was diagnosed with Gorham's disease.
Two years later, cervical imaging revealed involvement of the cervical vertebrae. External beam radiation was administered to the cervical spine and skull base. Nonetheless, the disease continued to progress and RG underwent spinal fusion for impending cervical instability. Repeated testing of the patient's Vitamin D, parathyroid hormone, and calcium levels were not altered throughout the disease process.
Two years later, a CT scan revealed progression of his disease with bony erosion and demineralization of his cervical spine and maxillofacial region, including near complete osteolysis of the mandible, platybasia, demineralization of the occipital bone, tympanic and mastoid portions of the temporal bone, greater wing of the right sphenoid bone, hypoglossal canal, and bilateral maxillary involvement (Figure 4). Seven years later, CT scan showed dramatic osteolytic defects of the right parietal, sphenoid, and right temporal bones as well as the posterior cervical spine (Figure 5).
The disease also progressed to involve the clavicle and ribs. In addition to the patient's spinal fusion and external beam radiation therapy, he was also treated with intravenous calcitonin and bisphosphonates. Although these treatment modalities may have slowed the progression of our patient's disease, they were not curative.
IMAGING AND RADIOLOGY
DISCUSSION
Gorham's disease is defined by osteolysis that can progress to complete dissolution of involved bone.14 The disease is usually monocentric, with contiguous involvement of adjacent bones.3 It can involve multiple skeletal regions, but rarely involves the facial skeleton. It usually affects patients in their second and third decades of life with an average age at diagnosis of 33 years old. Our case represents a pediatric patient with extensive axillofacial and skull base involvement. Both the age of the patient and location of disease are exceptionally rare manifestations of GD.
To the best of our knowledge, since 1928, there have only been three other cases of Gorham disease in the literature involving the mandible of a male patient younger than ten years of age. No regeneration has ever been observed following the osteolysis. GD seems to progress through two phases. The first phase consists of active bone resorption and contiguous spread to adjacent bone. Clinically, this may manifest as pathologic fractures, and may be the etiology of difficulties with speech, mastication, and a prominent facial deformity, all expressed in our patient. His facial deformity was particularly noticeable. Latent GD represents the second phase of massive osteolysis. During this quiescent phase, the haversian structure of the bone undergoes fibrosis. The duration of these stages is unpredictable and may last months to years. The lack of new bone formation is a trademark of GD.
The diagnosis of GD is based on clinical, histologic, and molecular features. Diagnosis is difficult and requires exclusion of neoplastic, inflammatory, infectious, and endocrinologic disease. The etiology of the massive osteolysis is still unknown. The lack of a standard treatment regimen for patients suffering from GD correlates with its widely disputed pathogenesis. The range of treatment consists of either surgery and radiation therapy, or medical therapy. The latter therapy is more disputed for the lack of a strong correlation with disease remission in affected patients. Early irradiation of the affected region has been observed to induce remission in some patients. Close observation for spinal instability is critical as these patients may commonly require surgical stabilization. Proper protection such as helmets may be needed. Medical therapy has been used in refractory cases and consists of bisphosphonates, calcitonin, calcium, vitamin D, and alpha-2b interferon therapy. Surgical treatment of disease involving the skull base and maxillo-facial regions may not be an option given the potential post-surgical morbidity. According to Escande, treatment of GD in the maxillofacial location is local resection of the involved bone in an attempt to stop progression of the disease. Although GD is an insidious progressive disease,
spontaneous regression has been described.8 Involvement of the cervical and thoracic spine resulting in persistent chylothorax is the usual cause of death in GD patients. Due to the rarity of pediatric GD, there is limited information on specific treatment options in pediatric patients.
The radiographic findings associated with the massive osteolysis in our patient were particularly dramatic. Near complete resorption of the mandible occurred. The ability to cause complete bone resorption has led to GD to be known as vanishing bone disease or phantom bone disease (Figures 4 and 5). Johnson and McClure divided the radiographic features of Gorham's disease into early intraosseous and later extraosseous stages. Early disease portrays patchy osteoporosis with multiple intramedullary and subcortical radiolucencies. In the later phase, disruption of the cortex leads to destruction and aggressive resorption of the bone. The spread of disease is contiguous with no apparent anatomical boundaries. There is insufficient literature describing the use of bone scans in GD and the few reports that have described their utility are not consistent. CT and MRI scanning is the imaging modality of choice for following the progression of GD.
CONCLUSSION
Gorham's disease is a complex disorder hallmarked by progressive osteolysis of unknown etiology that rarely manifests in the maxillo-facial region. To the best of our knowledge this case represents the fourth known case of Gorham's disease in a pediatric patient with mandibular involvment.
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2004;62:225-230.
GRANULAR CELL TUMOR OF THE BREAST
ABSTRACT
Introduction
A granular cell tumor involving the breast parenchyma was first described by Abrikossoff in 1931. Localization of this lesion to the breast is very rare, accounting for between 5% and 15% of all granular cell tumor cases. We present this case because of the rarity of this tumor. It is frequently confused with breast carcinoma on clinical and radiological examination, and its diagnosis can therefore be challenging for clinicians, radiologists and pathologists.
Case presentation
We report the case of a 32-year-old Moroccan woman who presented with a palpable mass in her right breast. Mammography and ultrasound examination revealed a heterogeneous, irregular and poorly limited mass, located at the union of the outer quadrants of her right breast. The mass was in contact with her latissimus dorsi and suspicious for malignancy. A histological examination combined with immunohistochemical study revealed it to be a granular cell tumor.
Conclusion
Although a granular cell tumor of the breast is a rare breast neoplasm, it should be considered in the differential diagnosis of benign and malignant lesions. Pathologists should bear in mind a granular cell tumor when examining material containing cells with abundant granular cytoplasm to avoid misdiagnosing breast carcinoma, which could lead to unnecessary surgery.
INTRODUCTION
A granular cell tumor (GCT) is an uncommon neoplasm that was first alluded to by Weber in 1854. It was fully described by Abrikossoff in 1926, who suspected a myogenic origin and therefore termed it a granular cell myoblastoma. However, because of S-100 protein positivity and the similarity of the tumor cells to Schwann cells, researchers proposed that the tumor originated from the Schwann cells; the exact histogenesis of this tumor is still unknown. It typically arises in the tongue but it may occur at any site and at any age, and can be multifocal. A GCT involving the breast parenchyma was first described by Abrikossoff in 1931. Localization of this lesion to the breast is very rare, accounting for between 5% and 15% of all GCT cases. Although GCT is a well-established entity, it is frequently confused on clinical and radiological examination with breast carcinoma. Its diagnosis may be a challenge for clinicians, radiologists and pathologists.
We report a case of a GCT of the breast mimicking carcinoma on mammography and ultrasonography. The diagnosis was made by histological examination. Through this observation, we discuss the radio-clinical, histopathological and therapeutic aspects of this rare tumor, as well as outcomes.
CASE PRESENTATION
We report the case of a 32-year-old Moroccan woman who presented with a palpable mass in her right breast of two year's duration. She had no personal or family history of malignancy. A physical examination showed a 1.5cm firm, painless mass located at the union of the outer quadrants of her right breast, without any alterations to her skin or axillary lymph nodes.A mammogram revealed a dense mass with ill-defined borders. An ultrasound demonstrated a 17mm hypoechoic, heterogeneous, irregular and poorly limited mass, located at the union of the outer quadrants of her right breast, in contact with her latissimus dorsi. The mass was suspicious for malignancy (Figure 1).On gross examination, the tumor was 2cm at its greatest diameter, whitish and had ill-defined borders. Microscopic examination revealed a benign tumor composed of compact nests of polygonal cells with well-defined cell borders that contained granular eosinophilic cytoplasm, and small, uniform, round nuclei without nuclear pleomorphism or mitotic activity (Figures 2 and 3). An immunohistochemical analysis showed positive staining for S-100 protein (Figure 4). The cells were negative for cytokeratins and cluster of differentiation (CD) 163. Based on these data, the diagnosis of GCT was confirmed.
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Figure 1. Ultrasound demonstrated a 17mm hypoechoic, heterogeneous, irregular and poorly limited mass, suspicious for malignancy.
Figure 2. Microscopic appearance: the tumor was composed of compact nests of polygonal cells with well-defined cell borders. Hematoxylin and eosin stain; original magnification ×100.
Figure 3. Microscopic appearance: tumor cells contained granular eosinophilic cytoplasm, and small, uniform, round nuclei without nuclear pleomorphism or mitotic activity. Hematoxylin and eosin; original magnification ×200.
Figure 4. Immunohistochemical study showed reactivity for S-100 protein. Original magnification ×100.
DISCUSSION
A GCT is an uncommon tumor that may arise throughout the body. The most common anatomical organ site of origin is the tongue, followed by soft tissues. GCT of the breast accounts for between 5% and 15% of all GCT cases. It occurs in a wide range of ages, from teenagers to the elderly, most commonly in women between 30 and 50 years of age, and especially in women of African-American origin. However, some cases of GCTs of the breast have been described in men.
This tumor usually presents as a firm and painless mass, usually well circumscribed, and generally mobile. Several cases have been reported with poorly circumscribed masses that may be fixed to the pectoral muscle, mimicking a malignant lesion. Skin involvement, including thickening, tethering, dimpling and retraction, has been described. Usually the tumor is solitary, but multiple (multifocal) lesions have been reported in 5.4% to 17.6% of cases. In our patient, the tumor was poorly limited and was in contact with her latissimus dorsi.
GCTs of the breast arise from intra-lobular breast stroma. They display no side preference. They occur more frequently in the upper inner quadrant of the breast, in contrast to breast carcinoma, which is more usually located in the upper outer quadrant. This reflects the course of the supraclavicular nerve, which innervates the breast skin. However, a case analysis elicited a wide variety of locations, including the upper outer quadrant, the upper inner quadrant, the axillary tail, the midline, the nipple and the subareolar region.
The histogenesis of GCT remains controversial and its etiology undiscovered. When Abrikossoff first described the tumour type he postulated that they originate in the skeletal muscle. Chung and Work went on to suggest a smooth muscle origin. Then it was thought that they derive from fibroblastic or undifferentiated mesenchymal or histiocytic cells and Ulrich et al. showing evidence of histiocytic origin. Additionally, immunohistochemical profiling suggests that they are unlikely to be of muscle (because of negativity for alpha-smooth muscle actin) or epithelial (because of negativity for keratin or epithelial membrane antigen) origin. Later, researchers proposed that the tumors originate from the Schwann cells because of their S-100 protein positivity and the similarity of the tumor cells to Schwann cells.
The presentation of GCTs of the breast on diagnostic imaging is variable. On mammography, it has often been described as a small (<3cm) lesion, ranging from a round, well-circumscribed mass, to an indistinct or spiculated lesion lacking calcifications, difficult to distinguish from carcinoma. On ultrasound, it can present as a solid, poorly marginated lesion, with marked posterior shadowing, suggestive of carcinoma, or as a more benign-appearing well-circumscribed solid mass.
A gross section usually shows a firm or hard, homogenous, grayish-white to yellow tumor, measuring generally 3cm or smaller, but tumors measuring up to 6cm have been reported. Most of the tumors appear to be well circumscribed, but other examples have ill-defined borders and may infiltrate, as in our case, into the surrounding tissues, particularly fibrous tissue, adipose tissue and the pectoralis major muscle. These features mimic malignant growth patterns and give the impression of scirrhous carcinoma.
On clinical and radiographic examination, it is impossible to establish a definitive diagnosis of GCT of the breast without a biopsy. Sonographically guided percutaneous biopsy of the lesion is well established as the diagnostic procedure of choice for histopathology sampling. On microscopy, the tumor is well circumscribed but may have infiltrative margins, as noted in our case. The cells are arranged in nests and sheets. They are generally uniform, large, bland and polygonal. However, rarely they may be round or spindle-like in shape. They have distinct borders and abundant granular eosinophilic cytoplasm, from which this tumor derives its name. The granular change is caused by cytoplasmic accumulation of lysosomes. Nuclei are small, centrally located and hyperchromatic with one or two nucleoli. They do not display mitoses, pleomorphism, nuclear multiplicity or atypia. Multi-nucleation and rare mitotic features may be seen, but these features should not be interpreted as evide\nce of malignancy. Variable amounts of collagenous stroma are present. Histochemical analysis confirms whether the granules are diastase resistant and Periodic acid-Schiff positive. The definitive diagnosis of GCT is only possible with immunohistochemical examination. The tumor cells are strongly immunoreactive to S-100 protein. They will not show staining for cytokeratins, epithelial membrane antigen or mucin. The cells were reported to be positive for CD68, carcinoembryonic antigen and vimentin in some cases in the literature. In our presented case, the description of the pathological features was supported by immunohistochemistry: S-100 protein positivity and cytokeratin negativity.
While the majority of GCTs behave in a benign manner, occasional malignant cases have been described (less than 1% of all GCTs, including mammary lesions, are malignant). The distinction between benign and malignant GCTs was proposed by Le et al and Adeniran et al., and included the criteria of necrosis, spindling, vesicular nuclei with large nucleoli, increased mitotic activity (more than two mitoses per 10 high power field at ×200 magnification), high nuclear to cytoplasmic ratio, and nuclear pleomorphism. These criteria classify GCT by histology into atypical (when two of these six criteria are present) and malignant (when three or more of these six criteria are met).
GCTs should be distinguished from mammary carcinoma, particularly scirrhous carcinoma and apocrine carcinoma. The difference between GCT and granulomatous inflammatory reaction or a histiocytic tumor is negativity for histiocyte-associated antigens, although reactivity for CD68 has been described in a GCT. GCTs must be distinguished from metastatic neoplasms of the breast that have oncocytic or clear cell features, such as renal carcinoma, malignant melanoma and alveolar soft part sarcoma.
Wide local excision with free margins is the treatment of choice. Subtotal excision may lead to local recurrences. Direct invasion of an axillary lymph node by a GCT of the breast that arose in the axillary tail has been reported.
CONCLUSION
This case illustrates that, although GCT of the breast is a relatively rare breast neoplasm, it should be considered in the differential diagnosis of benign and malignant lesions. Pathologist should bear in mind GCTs when examining material containing cells with abundant granular cytoplasm to avoid misdiagnosing breast carcinoma, which could lead to unnecessary surgery.
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EMPHYSEMATOUS CYSTITIS PRESENTING AS SEVERE CONFUSION AND ABDOMINAL PAIN: TWO CASE REPORTS
ABSTRACT
Introduction
Emphysematous cystitis is a very rare complicated urinary tract infection characterized by air in the bladder wall.
Case presentation
We report two clinical cases of emphysematous cystitis of an 83-year-old Caucasian woman with diabetes mellitus and a 78-year-old Caucasian man with no past medical history. They presented with severe confusion and abdominal distension. Emphysematous cystitis was diagnosed in time with a thorough physical examination, urine analysis and computed tomography. The patients were successfully treated with antibiotic therapy and bladder drainage.
Conclusion
This rare disorder should be recognized in time and treated properly to guarantee survival.
INTRODUCTION
Emphysematous cystitis is a very rare urinary tract infection. Two cases presenting as severe confusion and abdominal pain are described. Patients should be diagnosed in time by performing a thorough physical examination and selecting accurate diagnostic tests.
CASES PRESENTATION
CASE ONE
An 83-year-old Caucasian woman was referred by her general practitioner to our emergency department due to agitation and hallucination. Because of pyuria and haematuria on dipsticks, the general practitioner started ciprofloxacin 5 days earlier. She had a medical history of Parkinson disease, atrial flutter, rheumatoid arthritis and diabetes mellitus.
A physical examination revealed severe confusion, dehydration, abdominal distension and suprapubic tenderness.
Blood analysis revealed prerenal acute kidney failure (creatinine 2.24mg/dL, urea 98mg/dL), signs of infection (C-reactive protein 30mg/dL, serum white blood count 17200/mm3) and a well-controlled glycaemia (glycated haemoglobin, A1c, 48mmol/mol).
Urine analysis disclosed leukocyturia, bacteriuria and 540 red blood cells per field.
Because of the abdominal findings, an abdominal ultrasonography was performed. This showed irregular thickening of her bladder wall with extensive intramural echogenic foci (Figure 1).
Figure 1. Ultrasound of the bladder demonstrating irregular thickening of the bladder wall with extensive intramural echogenic foci.
In addition, a computed tomography was performed. This showed intraluminal air and emphysematous changes of her bladder wall (Figure 2).
Figure 2. Computed tomography. Axial view showing intraluminal air and emphysematous changes of the bladder wall.
Parenteral ciprofloxacin was continued and a 16 Fr Foley catheter was inserted for optimal drainage. After catheterization, pneumaturia was observed. A retrograde cystography was performed to exclude fistulas. This showed air at the posterior surface of her bladder and an irregular bladder wall with delineated radiolucent areas (Figure 3).
Figure 3. Retrograde cystography revealing air at the posterior surface of the bladder and an irregular bladder wall with delineated radiolucent areas.
To exclude outlet obstruction, a uroflowmetry was done. This examination revealed a micturition of 44mL with a maximum flow of 4.5mL/second. The contribution of this examination was limited because of the low micturition volume. Post-mictional residue was 250mL (Figure 4).
Figure 4. Uroflowmetry revealing a micturition of 44mL with a maximum flow of 4.5mL/second.
Cystoscopy revealed inflammatory lesions, diverticula and trabeculation (Figure 5).
Figure 5. Cystoscopy revealing inflammatory lesions, diverticula and trabeculation.
After 3 days, Escherichia coli resistant to ciprofloxacin but sensitive to temocillin was cultured at more than 1×106 colony-forming units/mL, so intravenous antibiotics were switched to the latter.
After 10 days she recovered. Her bladder catheter was removed and antibiotics were stopped. A repeat urine culture was sterile and she was sent home with clean intermittent catheterization. Nitrofurantoin was prescribed in the prophylaxis of urinary tract infections. A follow-up consultation at 4 weeks was arranged for uroflowmetry control. This examination revealed bladder function recovery with no post-mictional residue, so clean intermittent catheterization was stopped (Figure 6).
Figure 6. Revealing a micturition of 217mL with a maximum flow of 20.9mL/second.
CASE TWO
A 78-year-old Caucasian man with an adenocarcinoma of the pancreas was hospitalized for a pancreaticoduodenectomy. He has no past medical history. Four days after surgery, the bladder catheter was removed with subsequent normal micturition. One week after the Whipple procedure, he became aggressive, had severe confusion and abdominal pain. Abdominal palpation revealed diffuse tenderness without peritoneal signs. Laboratory testing showed a serum white blood count of 20200/mm3. Urine analysis showed significant leukocyturia and bacteriuria without haematuria. Because of the disturbing abdominal examination, abdominopelvic computed tomography was performed. This revealed emphysematous changes in his bladder wall (Figures 7 and 8).
Figure 7. Computed tomography. Axial view showing multiple gas foci in a diffuse collection of gas within the thickened bladder wall.
Figure 8. Computed tomography. Coronal view revealing emphysematous changes in the bladder wall.
Intravenous ciprofloxacin was started and a bladder catheter was replaced. After catheterization, pneumaturia was not observed. After 3 days, more than 1×106 colony-forming units/mL E. coli sensitive to ciprofloxacin was cultured. His abdominal pain and confusion disappeared after 2 days. After 1 week of treatment with antibiotics and bladder catheterization, his bladder wall returned to normal on computed tomography scanning of his abdomen.
CONCLUSIONS
Emphysematous cystitis is a rare disorder characterized by air in the bladder wall and lumen. The triggers and cellular mechanisms of this complicated urinary tract infection are unclear. Gas in the bladder wall is probably produced by natural fermentation of glucose by bacteria.
Older women with uncontrolled diabetes are at highest risk of developing emphysematous cystitis. A 2:1 female to male predominance is noticed and two-thirds of reported cases are diabetics. However, this complicated urinary tract infection should not be overlooked in non-diabetic male patients as presented in the second case. Other risk factors comprise urinary tract obstruction, indwelling urinary catheters, chronic urinary tract infections, neurogenic bladder or immunosuppression.
Presentation can vary from asymptomatic to severe sepsis. More than half of patients present with abdominal pain, classic symptoms of acute cystitis and pneumaturia. Although pneumaturia appears to be a highly specific symptom, it is a rare patient complaint. However, pneumaturia was observed after bladder catheterization in the first case. Both patients presented with severe confusion. This atypical symptom should always be further investigated as it can be the only sign of severe illness.
Urine analyses and Gram staining are essential in managing patients with cystitis emphysematosa. Laboratory testing usually reveals positive urine cultures with E. coli or Klebsiella pneumoniae involving 80% of cases. In the first case accurate treatment was delayed because of E. coli resistance to ciprofloxacin.
The severity and extent of emphysematous cystitis is best defined by computed tomography. Because of the increased use of computed tomography, emphysematous cystitis is often incidentally diagnosed. Images are characterized by multiple gas foci in a diffuse collection of gas within the thickened bladder wall.
When performing ultrasonography or plain films of the abdomen, air surrounding the bladder can be seen in most cases. Cystoscopy may show submucosal emphysema and can be suggestive for bladder outlet obstruction. The impeded flow from the bladder to the urethra can be confirmed by uroflowmetry.
Cystography can show irregular thickening of the bladder wall, representing submucosal blebs of air. Furthermore, it is the most sensitive imaging modality to exclude other causes of pneumaturia like enterovesical fistula.
In most cases, this gas-forming infection can be treated with antibiotics and bladder catheterization for optimal drainage. Of course, underlying medical conditions and glycaemic levels in diabetics should be controlled as well. In case of haematuria that includes blood clots or necrotizing tissue, bladder irrigation or transurethral debridement might be necessary. Emphysematous cystitis seldom requires (partial) cystectomy.
If not diagnosed in time, cystitis emphysematosa may result in urosepticaemia and even in death in up to 7% of patients.
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INTRAOSSEOUS LIPOMA OF THE THIRD LUMBAR SPINE
ABSTRACT
Introduction
Intraosseous lipoma is a benign bone tumor, and the tumor occurs more frequently in the lower extremities. We present a very rare case of intraosseous lipoma occurring in the lumbar vertebral arch and spinous process.
Case presentation
A 54-year-old Japanese man presented with a three-month history of lumbar pain. Magnetic resonance imaging of the L3 vertebral arch and spinous process revealed high intensity on T1- and T2- weighted imaging, and it was suppressed on fat-suppression imaging and no enhancement showed on gadolinium contrast-enhanced imaging. Computed tomography imaging revealed an osteolytic change accompanied by marginal osteosclerosis in his third lumbar vertebral arch and spinous process, as well as a thinned and bulging bone cortex. An analgesic had been administered prior to his visit, but low back pain had persisted, so we performed curettage and filled the defect with hydroxyapatite bone. His low back pain was improved immediately after surgery, and no recurrence of tumor has been observed on computed tomography imaging as of three years postoperatively.
Conclusions
Symptomatic intraosseous lipoma of spine is very rare, but the patient may be surgically well-treated by curettage and reconstruction of the benign tumor.
INTRODUCTION
Intraosseous lipoma is a benign bone tumor, and the tumor occurs more frequently in the lower extremities. We present a very rare case of intraosseous lipoma occurring in the lumbar vertebral arch and spinous process, together with a discussion of the literature.
CASE PRESENTATION
A 54-year-old Japanese man presented to our university-affiliated hospital with a three-month history of lumbar pain. He was 167cm in height and weighed 58kg. An analgesic had been administered prior to his visit, but low back pain had persisted. His physical examination showed pressure pain and tapping tenderness at the third lumbar vertebral level, but no sensory or motor disorders of his lower extremities. His blood biochemistry showed no abnormalities and his medical history was non-contributory.
A plain radiography revealed the formation of a vertebral spur or narrowing of the intervertebral disc between L3 and L4 as an age-related change, but no instability was evident between vertebrae and no obvious abnormalities were evident. Magnetic resonance imaging (MRI) of the L3 vertebral arch and spinous process revealed high intensity on T1- and T2-weighted imaging (Figure 1A,B,C), and it was suppressed on fat-suppression imaging (Figure 1D) and no enhancement showed on gadolinium (Gd) contrast-enhanced imaging (Figure 1E). Computed tomography (CT) imaging revealed an osteolytic change accompanied by marginal osteosclerosis in his third lumbar vertebral arch and spinous process, as well as a thinned and bulging bone cortex (Figure 2). Hounsfield units (HU) of CT for the area at which the osteolytic change was observed was 87HU, a value approximating that of fatty tissue, and areas of ossification or calcification were observed.
Figure 1.Magnetic resonance imaging at the L3 vertebral arch and spinous process. (A) Sagittal T1-weighted image; (B) Sagittal T2-weighted image; (C) Axial T2-weighted image; (D) Sagittal fat suppression image; (E) Sagittal T1-weighted gadolinium contrast (+) image.
Figure 2.Computed tomography image revealing an osteolytic change accompanied by marginal osteosclerosis in his third lumbar vertebral arch and spinous process, as well as a thinned and bulging bone cortex.
Based on the above findings, although we suspected painful lipoma in the third lumbar vertebral arch and spinous process segment, we decided to perform a biopsy to confirm the diagnosis. Since a benign tumor was suspected, we planned to perform curettage of the tumor and to fill the defect with artificial bone.
The operation was performed under general anesthesia. The third lumbar vertebral arch was exposed, and when an area approximately 1cm × 1cm in the external lamina of the right vertebral arch was opened, a yellow tumorous lesion with a color and elasticity macroscopically similar to those of ordinary fatty tissue was observed. The tumorous lesion was curetted away as much as possible, hydroxyapatite bone filler paste (BIOPEX®; HOYA Corporation, Tokyo, Japan) was used to fill the defect and the external lamina of the vertebral arch was replaced. Intraoperative pathological findings included hyperplasia of adipose cells and blood vessels, a small amount of trabecular bone and adipose cells of different sizes. Intraosseous lipoma was therefore diagnosed (Figure 3).
Figure 3.Intraoperative pathological findings of hyperplasia of adipose cells and blood vessels, a small amount of trabecular bone and adipose cells of different sizes. (Hematoxylin and eosin ×4 magnification).
His low back pain was improved immediately after surgery, and no recurrence of the tumor has been observed on CT imaging as of three years postoperatively (Figure 4).
Figure 4.Computed tomography image three years after surgery.
DISCUSSION
Despite the abundance of adipose connective tissue in bone marrow, intraosseous lipoma is extremely rare; a search of PubMed using the keywords 'intraosseous lipoma' yielded 177 results. A review of the search results showed that intraosseous lipoma occurs more frequently in the lower extremities, particularly in the calcaneus and metaphysis of long bones. Campbell et al. Reported that lipomas occur most frequently in the calcaneus (32%), while Milgram found that lipomas occur most frequently in the metaphysis of the proximal femur (34%).
We performed a review of the literature on intraosseous lipoma involving the spine, identifying only 14 cases (Table 1); five cases (35%) occurred in the lumbar region, four (28%) in the sacral region, three (21%) in the cervical region, one (7%) in the thoracic region and one (7%) in the coccygeal region. A slight predominance towards the lumbar spine was seen compared with other regions. The lesion in our patient also occurred in the lumbar region. Intraosseous lipoma in the lumbar region might occur at the vertebral body or in the posterior element, with a slight predominance toward the vertebral body. However, our patient presented with the lesion in the posterior element.
Table 1. Spinal intraosseous lipoma reported in the literature
Even though intraosseous lipoma is a benign tumor that can be successfully treated with conservative treatment, surgery has been recommended for diagnostic confirmation, painful tumors, pathological fractures and malignant transformation. In our patient, low back pain persisted after conservative treatment, so we performed curettage and filled the defect with hydroxyapatite bone. Most cases of intraosseous lipoma have no pain, however, micro-movement of the periosteum of the L3 vertebral arch and spinous process may have caused our patient's pain. Subsequently, the low back pain of our patient was improved immediately after filling the curetted defect with hydroxyapatite, and no recurrence of tumor has been observed as of three years postoperatively.
CONCLUSIONS
Symptomatic intraosseous lipoma of spine is very rare, but the patient may be surgically well-treated by curettage and reconstruction of the benign tumor.
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