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PubMed2003

Clinical and pathological studies of familial amyotrophic lateral sclerosis (FALS) with SOD1 H46R mutation in large Japanese families.

We clarified the clinical and pathological aspects of familial amyotrophic lateral sclerosis (FALS) with SOD1 H46R heterozygous mutation in the Miyakonojo Basin, a region in southern Japan where the prevalence of ALS is 11.4 per 10(5) of the population. We studied 17 patients, including one autopsy case, in three FALS families with the mutation. The average age at disease onset in the families was 44.3+/-8.7 years, and the mean disease duration was 12+/-7.6 years, with a range of 6 to 30 years. Ten of 17 patients were unable to walk by the mean age of 56.4+/-12.2 years. The initial symptom was muscle weakness in the distal leg muscle in all patients. The autopsy findings of one FALS patient showed atrophy of lateral and anterior funiculi, decreased numbers of anterior horn cells, preserved posterior funiculus and absence of neuronal inclusion bodies. Percentages of mutant SOD1 protein measured by mass spectrometry were 14% in erythrocytes, 43% in the spinal cord, 47% in the iliopsoas muscle and 60% in the diaphragm. In this study, we confirmed that FALS with SOD1 H46R mutation showed uniform initial symptoms and slow disease progression with intra-familial variation of disease severity and that inclusion body formation is not essential in FALS with this mutation.

Adult↗

PubMed1997

Fulminating gas-forming psoas muscle abscess due to Klebsiella pneumoniae following a deep neck infection.

Psoas muscle abscess due to Klebsiella pneumoniae infection is rare. We report a 55-year-old diabetic man who presented with progressive back pain of 1 month's duration. The patient had undergone surgical drainage for a deep neck infection with K. pneumoniae 43 days previously. On the present admission, physical examination revealed tenderness over the anterior upper aspect of both thighs, and computed tomography showed pneumoretroperitoneum dissecting the bilateral iliopsoas muscles. Parenteral administration of antibiotics was started immediately. Due to the patient's poor health status, we opted for repeated computed tomographic and sonographic-guided percutaneous drainage rather than surgical drainage. Blood and pus cultures revealed only K. pneumoniae. The patient recovered without significant sequelae. This report stresses the risk of metastatic infections caused by K. pneumoniae, especially in diabetic patients. Our experience suggests that repeated percutaneous drainage is feasible in cases of severe iliopsoas abscess, especially when risks associated with surgery are high.

Drainage↗

PubMedDate not supplied

Childhood onset oculopharyngeal muscular dystrophy.

Oculopharyngeal muscular dystrophy is an inherited disorder, usually autosomal dominant, which typically becomes symptomatic during the fifth decade of life with slowly progressive ptosis and dysphagia; childhood onset has not been reported. A 13-year-old female of French-Canadian descent developed nasal speech and strabismus at 5 years of age; there was no family history of neuromuscular disease. Ptosis and mild facial and proximal muscle weakness were present by 9 years of age. Over the next 4 years, the patient developed dysphagia, palatal paralysis, weight loss, decreased ocular motility, scoliosis, shortness of breath, and obstructive apnea. Tracheostomy and gastrostomy were required. Creatine kinase and repetitive facial nerve stimulation were normal. Edrophonium testing was negative and electromyography revealed myopathic motor units in the iliopsoas muscle. A preponderance of type I fibers and scattered atrophic and angulated muscle fibers were present in 3 muscle biopsies. The clinical presentation and findings are consistent with childhood onset oculopharyngeal muscular dystrophy.

Adolescent↗

PubMed2000

[A family with oculopharyngeal muscular dystrophy with (GCG)9 expansion in which a sister had neck as well as proximal and her brother proximal lower limb muscle weakness].

We report a 58-year-old woman (patient 1) and her 60-year-old brother (patient 2) with autosomal dominant oculopharyngeal muscular dystrophy. Patient 1 first noticed blepharoptosis and neck weakness at age 55. On neurological examination, she showed bilateral blepharoptosis and weakness in the neck and upper proximal limbs. Serum creatine kinase (CK) level was slightly elevated. Her older brother first noticed blepharoptosis and lower limb weakness at age 51. On neurological examination, he showed bilateral blepharoptosis, slight ophthalmoparesis and bilateral iliopsoas muscle weakness. Serum CK level was normal. Esophageal fluoroscopy disclosed dysfunction of the constrictor pharyngeal muscles. Muscle biopsy of them showed myopathic changes with rimmed vacuoles. The (GCG)9 mutation in the poly (A) binding protein 2 gene was identified, which was the same as seen in the large French-Canadian kindred in Quebec in Canada. The clinical phenotype in patient 2 is similar to that of French-Canadian patients but it in patient 1 is different in distribution of muscle weakness.

DNA-Binding Proteins↗

PubMed2000

[A patient with recurrent uterine cervical cancer successfully treated by combined modality therapy including local injection of anticancer agents based on CDDP].

The patient was a 77-year-old woman who had undergone total abdominal hysterectomy and bilateral salpingo-oophorectomy following the diagnosis of uterine cervical cancer at the age of 57. A recurrent tumor was detected on the anterior surface of the right iliopsoas muscle and fine needle aspiration of the tumor revealed squamous cell carcinoma. A needle was inserted into the tumor under transabdominal ultrasound guidance, and anticancer agents, chiefly cis-diamminedichloroplatinum [II] (CDDP), were injected. The patient also received systemic chemotherapy (CAP therapy) and radiotherapy. Subsequently, computed tomography showed a 73% reduction in the size of the tumor. A mass 7 cm in diameter was still present on the anterior surface of the iliopsoas after treatment, but tumor markers fell to normal levels. The patient is presently receiving UFT (600 mg/day) as maintenance therapy. At 48 months after recurrence, there has been no increase in the tumor size or tumor marker levels, and the performance status is grade 0. In conclusion, a good outcome was achieved by local injection of anticancer agents combined with systemic chemotherapy and radiotherapy, and there were few side effects.

Aged↗

PubMed1996

Contribution of hind limb flexor muscle afferents to the timing of phase transitions in the cat step cycle.

1. In this investigation, we tested the hypothesis that muscle spindle afferents signaling the length of hind-leg flexor muscles are involved in terminating extensor activity and initiating flexion during walking. The hip flexor muscle iliopsoas (IP) and the ankle flexors tibialis anterior (TA) and extensor digitorum longus (EDL) were stretched or vibrated at various phases of the step cycle in spontaneously walking decerebrate cats. Changes in electromyogram amplitude, duration, and timing were then examined. The effects of electrically stimulating group I and II afferents in the nerves to TA and EDL also were examined. 2. Stretch of the individual flexor muscles (IP, TA, or EDL) during the stance phase reduced the duration of extensor activity and promoted the onset of flexor burst activity. The contralateral step cycle also was affected by the stretch, the duration of flexor activity being shortened and extensor activity occurring earlier. Therefore, stretch of the flexor muscles during the stance phase reset the locomotor rhythm to flexion ipsilaterally and extension contralaterally. 3. Results of electrically stimulating the afferents from the TA and EDL muscles suggested that different groups of afferents were responsible for the resetting of the step cycle. Stimulation of the TA nerve reset the locomotor step cycle when the stimulus intensity was in the group II range (2-5 xT). By contrast, stimulation of the EDL nerve generated strong resetting of the step cycle in the range of 1.2-1.4 xT, where primarily the group Ia afferents from the muscle spindles would be activated. 4. Vibration of IP or EDL during stance reduced the duration of the extensor activity by similar amounts to that produced by muscle stretch or by electrical stimulation of EDL at group Ia strengths. This suggests that the group Ia afferents from IP and EDL are capable of resetting the locomotor pattern generator. Vibration of TA did not affect the locomotor rhythm. 5. Stretch of IP or electrical stimulation of TA afferents (5 xT) during the flexion phase did not change the duration of the flexor activity. Stimulation of the EDL nerve at 1.8-5 xT during flexion increased the duration of the flexor activity. In none of our preparations did we observe resetting to extension when the flexor afferents were activated during flexion. 6. We conclude that as the flexor muscles lengthen during the stance phase of gait, their spindle afferents (group Ia afferents for EDL and IP, group II afferents for TA) act to inhibit the spinal center generating extensor activity thus facilitating the initiation of swing.

Afferent Pathways↗

PubMed1998

Primary iliopsoas abscess successfully treated by ultrasonographically guided percutaneous drainage.

We report a case of primary iliopsoas abscess successfully treated by ultrasonographically guided percutaneous drainage. A 56-year-old man presented at our hospital with lumbago, right-sided back pain, fever (temperature 38.5 degrees C) and chills. On physical examination, we found dark red skin, swelling, and tenderness localized at the right side at the back of his waist. Laboratory examination showed leukocytosis (white blood cell count 9700/mm3) with a leftward shift and elevated C-reactive protein (5.2 mg/dl). Ultrasonography (US), computed tomography (CT), and magnetic resonance imaging revealed a hypodense lesion in the right iliopsoas muscle extending to the subcutaneous tissue. About 50 ml of thick yellow pus was obtained by ultrasonographically guided aspiration drainage. A drain catheter was inserted in the abscess cavity. Laboratory findings improved and clinical symptoms abated rapidly after drainage. On the twenty-first day after drainage, US and CT showed that the abscess was no longer present. The patient was discharged after 32 days of hospitalization. As possible primary diseases causing iliopsoas abscess, such as digestive tract disease, tuberculosis, and osteomyelitis, were not found, we diagnosed the disease as primary iliopsoas abscess. Although surgical drainage has been performed in most reported cases of iliopsoas abscess, this case report shows that ultrasonographically guided percutaneous drainage is also effective for treating primary iliopsoas abscess if it is diagnosed early enough.

C-Reactive Protein↗

PubMed1995

Changes in hip muscles after above-knee amputation.

To learn about the changes appearing in hip muscles after an above-knee amputation, 3-dimensional reconstructions of the hip and thigh region of 12 patients with above-knee amputations were made based on transverse magnetic resonance images. In all patients, the amputations were done at least 2 years before the study and were necessitated by trauma or osteosarcoma. The results show that, at higher amputation levels, the geometry of the once-biarticular muscles was changed. The cleaved muscles (40%-60%) and the intact muscles (0-30%) at the amputated side were atrophied. The amount of atrophy of the intact muscles at the amputated side was related to stump length. To avoid an abduction contracture in 8 patients with amputations, the iliotibial tract was not fixed. In 4 of these 8 patients, a flexion contracture was visible. If the tract was not fixed, the hip extension torque of the gluteus maximus, which inserts into the tract, decreased. As a result, the risk of appearance of a flexion contracture increased because the strongest hip flexor (iliopsoas muscle) was not involved in the amputation. Abduction contracture could be avoided only if the hip adductors were fixed accurately, especially at higher amputation levels.

Adult↗

PubMed1979

Correlation between muscle shortening and derangement of the hip joint in children with spastic cerebral palsy.

From clinical studies on hip derangement and measurement of muscle shortening around the hip joint in 244 patients with spastic cerebral palsy, we found that the incidence of subluxation was 17.0% and that of dislocation was 8.2%; there was no dislocation in ambulatory spastics and the incidence of hip derangement was high in non-ambulatory patients; significant shortening of the adductors was closely correlated with hip derangement; the combination of damaged flexors, hamstrings and adductors led to hip derangement; surgical intervention in the adductors, the flexors (particularly the iliopsoas muscle) and the hamstrings should be considered in the treatment of advanced hip derangement in patients with cerebral palsy.

Adolescent↗

PubMed1996

[A 63-year-old woman with muscle weakness, myotonia, and parkinsonism].

We report a 63-year-old woman who presented myotonia and parkinsonism. The patient was well until 15 years of the age when she noted that the ring finger of her left hand at times flexed when she did not intend to do so. She noted weakness in her left upper extremity at the age of 40, and difficulty in relaxing her hand grip at 45. She had an onset of tremor in her right foot at age 50, which was followed by difficulty in gait and hand writing. She was admitted to Juntendo University Urayasu Hospital when she was 63-year-old. Her mother, two sisters, and a son were affected with similar muscle weakness and myotonia. Although some of them developed stooped posture in the late stage of the disease, none of them had overt parkinsonism. General physical examination was unremarkable. Neurologic examination revealed an alert and oriented woman with some recent memory loss. She had bilateral ptosis, facial weakness, and a masked face. Myerson's sign was present. Her speech was small and monotonous. The sternocleidomastoid muscles were markedly atrophic and weak. The remaining of the cranial nerves were intact. She walked in small steps with freezing with support. She showed bradykinesia, retropulsion, and resting tremor in her right leg. Slight distal dominant weakness was noted in both upper and lower extremities more on the left. No cerebellar signs were noted. Muscle stretch reflexes were within normal limits in the upper extremities and diminished in the lower limbs. Sensation was intact. Routine laboratory findings were unremarkable. Cranial CT scan and MRI revealed slight cortical atrophy and leukoaraiosis. She responded to levodopa and she became able to walk by herself. She was transferred to another hospital one month after her admission. She had several bouts of airway obstruction with one episode of respiratory arrest. She expired six month after the transfer. The patient was discussed in a neurological CPC, and the chief discussant arrived at the conclusion that this patient suffered from myotonic dystrophy and Parkinson's disease which set in later years. Postmortem examination on the iliopsoas muscle revealed uneven muscle fiber diameters, central nuclei, and type 1 fiber predominance; the pathologic finding was consistent with myotonic dystrophy. The substantia nigra showed marked cell loss and Lewy bodies in the remaining neurons. The finding was consistent with Parkinson's disease. In myelin stain, diffuse myelin pallor was noted in the cerebral white matter which was the pathologic substrate of leukoaraiosis in this patient. Combination of these two disorders have never been reported in the literature to our knowledge. It appears to be that the coincidence is just a by-chance phenomenon, but it seems interesting to note that accelerated aging process appears to be present in both myotonic dystrophy and Parkinson's disease.

Diagnosis, Differential↗

PubMed2004

Interpretation of a 3,4-methylenedioxymethamphetamine (MDMA) blood level: discussion by means of a distribution study in two fatalities.

The amphetamine derivative 3,4-methylenedioxymethamphetamine (MDMA, "Ecstasy" is a currently used or abused designer drug and fatalities are frequently encountered in forensic practice. However, the question remains open whether an MDMA blood level can be toxic or even potentially lethal. In order to provide insight in the interpretation of a detected MDMA concentration, the distribution of MDMA and its metabolite 3,4-methylenedioxyamphetamine (MDA) in various body fluids and tissues was studied and discussed in two different fatalities. Apart from peripheral blood samples (such as femoral and subclavian blood), various blood samples obtained centrally in the human body and several body fluids (such as vitreous humour) were examined. In addition, various tissues such as cardiac muscle, lungs, liver, kidneys, and brain lobes were analysed. In contrast to the peripheral blood levels, high MDMA and MDA levels were found in cardiac blood and the majority of the organs, except for the abdominal adipose tissue. The high concentrations observed in all lung lobes, the liver and stomach contents indicate that post-mortem redistribution of MDMA and MDA into cardiac blood can occur and, as a result, blood sampled centrally in the body should be avoided. Therefore, our data confirm that peripheral blood sampling remains "the golden standard". In addition, a distinct difference in peripheral blood MDMA concentrations in our two overdose cases was established (namely 0.271 and 13.508 microg/ml, respectively). Furthermore, our results suggest that, if a peripheral blood sample is not available and when putrefaction is not too pronounced, vitreous humour and iliopsoas muscle can be valuable specimens for toxicological analysis. Finally, referring to the various mechanisms of death following amphetamine intake, which can result in different survival times (e.g. cardiopulmonary complications versus hyperthermia), the anatomo-pathological findings and the toxicological results should be considered as a whole in arriving at a conclusion.

3,4-Methylenedioxyamphetamine↗

PubMed1992

Bilateral ankylosis of the hips following heterotopic ossification of the ilio-psoas in a child.

A case of bilateral ankylosis of the hip in a 3-year-old child due to heterotopic ossification of the iliopsoas following a long period of coma is presented. A review of the literature has failed to reveal a similar case with such extensive heterotopic ossification. The patient was treated successfully by surgical excision of the heterotopic bone which was extra-articular and extra-capsular and involved only the iliopsoas muscles.

Ankylosis↗

PubMed1992

Reconstruction of the proximal femur with a vascular pedicle iliac crest graft after tumour resection.

A useful method of reconstruction of the proximal femur after tumour resection is described. An anterior iliac crest bone graft with an intact vascular supply based on the deep circumflex iliac vascular bundle is used to fill the bone defect. It is swung down to the femur behind the iliopsoas muscle and locked into place in the femur. Sixteen cases of locally aggressive tumours have been resected and reconstructed by this method. Follow-up for five to eight years shows good reconstitution of the femur and hip function without recurrence.

Adult↗

PubMed1982

Ruptured abdominal aortic aneurysms presenting as radicular compression syndromes.

Two patients are described: one with an aneurysm of the infrarenal aorta and common iliac artery that ruptured posteriorly into the iliac vein, the other with an aneurysm of the distal abdominal aorta that ruptured posteriorly into the iliopsoas muscle. Both patients had symptoms compatible with a radicular compression syndrome. Ruptured aneurysm of one of the major abdominal arteries should be considered in the differential diagnosis of affections of the lumbosacral neural outflow, because immediate operation can be life-saving.

Aorta, Abdominal↗

PubMed1988

The lateral branches of the common iliac artery.

The description of the common iliac artery and its branches varies significantly in various anatomic references. The results of the observations of several authors are discussed. A study of 143 cadavers in which the branches of the common iliac arteries are investigated is reported. In this study 93 cases (65%) showed no branch from the common iliac artery. In 50 cases (35%) a lateral branch was found. Out of these 50, 18 were given off from the right side, 16 from the left and 16 from both sides. The average diameter of the branch lumen was about 3 mm. The branches supplied the iliopsoas muscle (in addition to the iliolumbar artery), occasionally the ureter, lymphnodes or kidney. In operative procedures near the bifurcation of the abdominal aorta it is necessary to look for lateral branches of the common iliac artery.

Female↗

PubMed2004

Intra-abdominal gout mimicking pelvic abscess.

Gout is the most common crystal-induced arthritis. Gouty tophi typically deposit in the extremities, especially toes and fingers. We present an unusual case of intrapelvic tophaceous gout in a patient suffering from chronic gouty arthritis. CT and MRI of the abdomen and pelvic cavity disclosed calcified gouty tophi around both hips, and a cystic lesion with peripheral enhancement in the pelvic cavity along the course of the iliopsoas muscle. The intra-abdominal tophus mimicked pelvic abscess.

Abdomen↗

PubMedDate not supplied

Ileocecal actinomycosis: a case report.

A 51-year-old male with a clinical history of abdominal pain and bloody diarrhea underwent spiral computed tomography of the abdomen with intravenous contrast medium enhancement. The findings were an inflammatory mass with cystic and solid characteristics arising from the ileocecal region and extending to the peritoneum and right iliopsoas muscle, with adjacent lymphadenopathy. Histology after surgical resection confirmed the diagnosis of actinomycosis lesions in perienteric fat.

Actinomycosis↗

PubMedDate not supplied

Percutaneous decompression of an iliopsoas hematoma.

Spontaneous hematoma in the iliopsoas muscle is a serious complication of anticoagulant therapy. It causes severe pain and often results in loss of function of the femoral nerve. We present two cases in which percutaneous decompression of the hematoma was successfully performed to relieve intractable pain and to support functional restoration of the femoral nerve.

Aged↗