Sparganosis, an infection due to the plerocercoid of Spirometra erinacei, are found worldwide but the majority of cases occur in East Asia including Korea. This report is on a recurred case of sparganosis in the subcutaneous tissue of the right lower leg 1 year after a surgical removal of a worm from a similar region. At admission, ultrasonography (USG) of the lesion strongly suggested sparganosis, and a worm was successfully removed which turned out to be a sparganum with scolex. Since sparganum has a variable life span, and may develop into a life-threatening severe case, a patient once diagnosed as sparganosis should be properly followed-up for a certain period of time. Although imaging modalities were useful for the diagnosis of sparganosis as seen in this case, serological test such as ELISA should also be accompanied so as to support the preoperative diagnosis.
Spirometra erinacei; sparganum; sparganosis; ultrasonography
Sparganosis is an infection with a parasitic tapeworm larva that occurs by eating infected foods or drinking contaminated water. The larvae can migrate to a tissue or muscle in the chest, abdominal wall, extremities, eyes, brain, urinary tract, pleura, pericardium, spinal canal, or scrotum. Herein, we report a 5-month old infant with scrotal sparganosis who was initially suspected to have a scrotal inflammatory mass with a history of applying raw frog meat into the umbilicus. Preoperative ultrasound examinations and computed tomography (CT) scanning misdiagnosed the mass as a scrotal teratoma. The scrotal mass was surgically removed, and the histopathology proved it to be scrotal sparganosis. This case displays the youngest patient ever reported with scrotal sparganosis, and the first description of CT characteristics of scrotal sparganosis. A detailed medical history is necessary for patients with scrotal masses suspected of sparganosis. In addition, ultrasound and CT examinations are helpful to rule out other causes of a scrotal mass.
Sparganum; sparganosis; scrotum; computed tomography; ultrasound
Sparganosis is caused by a larval tapeworm of the genus Spirometra, which commonly invades subcutaneous tissue, but less frequently invades muscle, intestines, spinal cord, and the peritoneopleural cavity. The authors managed a female patient who presented with a recurrent pericardiopleural effusion and peripheral eosinophilia. The anti-sparganum-specific IgG serum level was significantly higher than normal control levels. In this patient, sparganosis was caused by the ingestion of raw frogs in an effort to control her thyroid disease. The recurrent pericardiopleural effusion and peripheral eosinophilia were controlled by 3 consecutive doses of praziquantel (75 mg/kg/day). The patient is doing well 4 years after presentation. Sparganosis should be considered a rare, but possible cause of recurrent pericardial effusion and peripheral eosinophilia. Immunoserologic testing using enzyme linked immunosorbent assays can be helpful in diagnosing human sparganosis, especially in cases without a subcutaneous lump or mass. Praziquantel is an alternative treatment for sparganosis in surgically-unresectable cases.
Sparganosis; Pericardial effusion; Praziquantel
Sparganosis is a rare, parasitic infection that is caused by the plercercoid tapeworm larvae of the genus Spirometra. Sparganosis is transmitted by ingestion of larvae-containing water or intermediate hosts, such as raw amphibians, and usually presented with a subcutaneous nodule in the abdominal wall, extremities, and genital organs. Among the various involved organs, the breast is a seldom encountered site for sparganosis. However, breast sparganosis has clinical importance, since it is generally presented with an indolent palpable mass that mimics malignancy, even without evidence of inflammation. Herein, we report a case of breast sparganosis that was detected incidentally by FDG PET/CT during staging work ups in a patient with gall bladder cancer.
Human sparganosis; 18F-FDG; PET/CT; Breast
Sparganosis mansoni is a parasitic disease caused by the larva of Spirometra mansoni. It occurs worldwide, but only a few patients show pulmonary involvement. Here, we present a case of pulmonary sparganosis mansoni in a non-endemic region. A 32-year-old Chinese woman presented with intermittent bloody phlegm, peripheral blood eosinophilia, and migratory patch shadows in both lungs. She had been misdiagnosed with eosinophilic pneumonia. She had a history of eating raw frogs, and the sparganum mansoni antibody was positive in both her blood and bronchoalveolar lavage fluid. Several sparganum mansoni were found in a frog sample that the patient provided. Consequently, she was diagnosed with pulmonary sparganosis mansoni. After two oral courses of praziquantel were administered, her symptoms and radiological lesions improved significantly. To our knowledge, this is the first case of pulmonary sparganosis mansoni occuring in Shanghai. Oral praziquantel is effective for the treatment of sparganosis mansoni, although its course of therapy may need to be repeated.
Lung; sparganosis mansoni; diagnosis; treatment
Sparganosis is a rare parasitic infection caused by the plerocercoid tapeworm larva of the genus Spirometra.
We report the case of a 67-year-old man with a mass over the anteromedial surface of the proximal extremity of the tibia. We surgically excised a bursa containing Spirometra larvae.
Sparganosis is a rare parasitic infection. We found no cases of lower extremity sparganosis combined with bursitis reported in the literature.
Purposes and Clinical Relevance
Sparganosis should be considered in the differential diagnosis of soft tissue tumors, especially among patients who frequently have consumed mountain water or raw snakes or frogs.
Sparganosis is a rare parasitic disease caused by migrating plerocercoid tapeworm larva of the genus Spirometra. Infection in humans is mainly caused by the ingestion of raw or inadequately cooked flesh of infected frogs, snakes, and chickens. Here, we report a rare case of a 45-year-old man who was admitted to our hospital with left lower chest pain. The chest radiograph and computed tomography (CT) scan revealed localized pleural effusion in the left lower lobe; further, peripheral blood eosinophilia and eosinophilic pleural effusion were present. Percutaneous catheter drainage was performed, which revealed long worm-shaped material that was identified as a sparganum by DNA sequencing. The patient showed clinical improvement after drainage of the sparganum. This study demonstrates the importance of considering parasitic diseases in the differential diagnosis of eosinophilic pleural effusion.
Sparganosis; eosinophilic pleural effusion; DNA sequencing
Sparganosis is caused by the invasion of Spirometra sparganum into various tissues/organs. Subcutaneous sparganosis can be diagnosed by biopsy, while visceral/cerebral sparganosis is not easy to be diagnosed. The diagnosis depends largely on the detection of specific anti-sparganum antibodies. The specificity of the ELISA could be increased by using S. mansoni sparganum excretory–secretory (ES) antigens, but it also had the cross-reactions with sera of patients with cysticercosis or paragonimiasis. The aim of this study was to identify early specific diagnostic antigens in S. mansoni sparganum ES proteins.
The sparganum ES proteins were analyzed by two-dimensional electrophoresis (2-DE) and Western blot probed with early sera from infected mice at 14 days post-infection. The immunoreactive protein spots were characterized by MALDI-TOF/ TOF-MS.
A total of approximately 149 proteins spots were detected with isoelectric point (pI) varying from 3 to 7.5 and molecular weight from 20 to 115 kDa and seven protein spots with molecular weight of 23-31 kDa were recognized by the infection sera. Three of seven spots were successfully identified and characterized as the same S. mansoni protein (cysteine protease), and the proteins of other 4 spots were not included in the databases.
The cysteine protease from S. mansoni ES proteins recognized by early infection sera might be the early diagnostic antigens for sparganosis.
Spirometra mansoni; Plerocercoid; (sparganum); Excretory-secretory; Diagnosis
Antigenic components in the crude extracts of Spirometra mansoni plerocercoid were analyzed in early experimental infections and in IgG subclass observed in clinical sparganosis. By IgG immunoblot, sera obtained serially from experimental mice, fed 5 spargana each, were reacted with the crude extracts. Protein bands at 36-26 kDa and 103 kDa showed positive reactions since two weeks after infection. In a differential immunoblot, in which a monospecific antibody against sparganum chymase at 36 kDa was pre-treated, the reactions at 36-26 kDa disappeared, indicating that the sparganum chymase and its degradation products invoked IgG antibody reactions. When 69 patients sera of human sparganosis were examined for their IgG subclass responses, IgG4 levels showed the highest reaction which was followed by IgG1. The IgG4 antibody also reacted mainly with 36-31 kDa protease. These results indicate that 36 kDa chymase of S. mansoni plerocercoid is the main antigenic component inducing IgG antibody response in early stage of experimental sparganosis and for specific IgG subclass reactions in human sparganosis.
Spirometra; Sparganum; Sparganosis; Antigens; Antibodies; Immunoblotting
Central nervous system (CNS) sparganosis is a rare parasitic infestation caused by ingestion of the raw or inadequately cooked snakes or frogs. Sparganum is well known for its ability of migrating though the tissue, therefore, it can cause various neurological symptoms if it involves neurological systems. A 51-year-old male patient visited our department of neurosurgery complaining of the motor weakness and radiating pain on both upper extremities over 4 months. He had a history of ingesting raw snakes untill his late twenties. The magnetic resonance (MR) images of cervical spine revealed an intramedullary ill-defined enhancing lesion with the aggregated cysts in the upper cervical spinal cord. Under presumptive diagnosis of sparganosis, we took brain MR image. The brain MR images revealed the signal change in right fronto-temporal lobe suggesting the trajectory of parasitic migration via ventricular systems. He underwent a midline myelotomy and granuloma removal followed by the posterior laminoplasty. Pathologic findings showed inflammatory changes and necrosis with keratinized tissue suggesting the CNS sparganosis. We report an uncommon case of CNS sparganosis migrated from the brain to the spinal cord with literature review.
Central nervous system; Migration; Sparganosis; Spinal cord; Ventricle
Sparganosis is an infection with a larval Diphyllobothriidea tapeworm. From a rare cerebral case presented at a clinic in the UK, DNA was recovered from a biopsy sample and used to determine the causative species as Spirometra erinaceieuropaei through sequencing of the cox1 gene. From the same DNA, we have produced a draft genome, the first of its kind for this species, and used it to perform a comparative genomics analysis and to investigate known and potential tapeworm drug targets in this tapeworm.
The 1.26 Gb draft genome of S. erinaceieuropaei is currently the largest reported for any flatworm. Through investigation of β-tubulin genes, we predict that S. erinaceieuropaei larvae are insensitive to the tapeworm drug albendazole. We find that many putative tapeworm drug targets are also present in S. erinaceieuropaei, allowing possible cross application of new drugs. In comparison to other sequenced tapeworm species we observe expansion of protease classes, and of Kuntiz-type protease inhibitors. Expanded gene families in this tapeworm also include those that are involved in processes that add post-translational diversity to the protein landscape, intracellular transport, transcriptional regulation and detoxification.
The S. erinaceieuropaei genome begins to give us insight into an order of tapeworms previously uncharacterized at the genome-wide level. From a single clinical case we have begun to sketch a picture of the characteristics of these organisms. Finally, our work represents a significant technological achievement as we present a draft genome sequence of a rare tapeworm, and from a small amount of starting material.
Electronic supplementary material
The online version of this article (doi:10.1186/s13059-014-0510-3) contains supplementary material, which is available to authorized users.
We report here a case of inguinal sparganosis, initially regarded as myeloid sarcoma, diagnosed in a patient undergone allogeneic hematopoietic transplantation (HSCT). A 56-year-old male patient having myelodysplastic syndrome was treated with allogeneic HSCT after myeloablative conditioning regimen. At day 5 post-HSCT, the patient complained of a painless palpable mass on the left scrotum and inguinal area. Pelvic magnetic resonance imaging and computed tomography revealed suspected myeloid sarcoma. Gun-biopsy was performed, and the result revealed eosinophilic infiltrations without malignancy. Subsequent serologic IgG antibody test was positive for sparganum. Excisional biopsy as a therapeutic diagnosis was done, and the diagnosis of sparganosis was confirmed eventually. This is the first report of sparganosis after allogeneic HSCT mimicking myeloid sarcoma, giving a lesson that the physicians have to consider the possibility of sparganosis in this clinical situation and perform adequate diagnostic and therapeutic approaches.
Sparganum; sparganosis; myeloid sarcoma; hematopoietic stem cell transplantation; myelodysplastic syndrome
Sparganosis is a parasitic infection caused by the plerocercoid tapeworm larva of the genus Spirometra. Although the destination of the larva is often a tissue or muscle in the chest, abdominal wall, extremities, eyes, brain, urinary tract, spinal canal, and scrotum, intramuscular sparganosis is uncommon and therefore is difficult to distinguish from a soft tissue tumor. We report a case of intramuscular sparganosis involving the gastrocnemius muscle in an elderly patient who was diagnosed using ultrasonography and MRI and treated by surgical excision. At approximately 1 cm near the schwannoma at the right distal sciatic nerve, several spargana worms were detected and removed.
Spirometra sp.; sparganosis; intramuscular; gastrocnemius muscle; schwannoma
The authors report the case of a 46-year-old woman with cerebral sparganosis resulting from infection with a larva of Spirometra. Computed tomography and magnetic resonance imaging revealed a mass lesion with prominent perifocal oedema in the left parietal lobe. Advanced imaging pulse sequences, including MR spectroscopy and MR perfusion, were performed. During surgery for the removal of a granuloma, the parasite was discovered and excised. Following treatment, the patient's neurological deficits markedly improved.
A 29-year-old Korean woman visited the Department of Surgery in MizMedi Hospital with a palpable itching mass on the right breast that had existed for the past 7 months. She had no history to eat either frogs or snakes, but had the history of drinking impure water. Sonography revealed a serpiginous hypoechoic tubular structure associated with partial fat necrosis in breast parenchymal layer and subcutaneous fat layer. It also revealed oval cystic lesions. At operation, an ivory white opaque ribbon-like worm that measured 16.5 cm in length and 0.5 cm in width was extracted. Anti-sparganum specific serum IgG level in the patient's serum (absorbance = 0.71), measured by ELISA, was found to be significantly higher than those of normal controls (cut off point = 0.21). Sonography and ELISA appear to be helpful to diagnose sparganosis. Breast sparganosis is rarely found throughout the world.
sparganosis; breast; ultrasonography
Magnetic resonance imaging sequences such as diffusion and spectroscopy have been well studied in X-linked adrenoleukodystrophy, but no data exist on magnetic resonance perfusion imaging. Since inflammation is known to modulate the microcirculation, we investigated the hypothesis that changes in the local perfusion might be one of the earliest signs of lesion development. Twenty patients with different phenotypes of adrenoleukodystrophy and seven age-matched controls were evaluated between 2006 and 2011. Fluid attenuated inversion recovery, post-contrast T1-weighted and normalized dynamic susceptibility contrast magnetic resonance perfusion cerebral blood volume maps were co-registered, segmented when cerebral lesion was present, and normalized cerebral blood volume values were analysed using a Food and Drug Association approved magnetic resonance perfusion software (NordicICE). Clinical and imaging data were reviewed to determine phenotype and status of progression. All eight patients with cerebral adrenoleukodystrophy had an average 80% decrease in normalized cerebral blood volume at the core of the lesion (P < 0.0001). Beyond the leading edge of contrast enhancement cerebral perfusion varied, patients with progressive lesions showed an average 60% decrease in normalized cerebral blood volume (adults P < 0.05; children P < 0.001), while one child with arrested progression normalized cerebral blood volume in this region. In six of seven patients with cerebral adrenoleukodystrophy lesions and follow-up imaging (2–24 month interval period), we found progression of contrast enhancement into the formerly hypoperfused perilesional zone. Asymptomatic, adrenomyeloneuropathy and female heterozygote patients had no significant changes in cerebral perfusion. Our data indicate that decreased brain magnetic resonance perfusion precedes leakage of the blood–brain barrier as demonstrated by contrast enhancement in cerebral adrenoleukodystrophy and is an early sign of lesion progression.
MRI perfusion; demyelination; neuroinflammation; adrenoleukodystrophy; leukodystrophy
To prospectively compare 0.1 mmol/kg doses of gadobenate dimeglumine and gadopentetate dimeglumine for contrast-enhanced MRI of brain lesions at 3 Tesla (T).
Materials and Methods
Forty-six randomized patients underwent a first examination with gadobenate dimeglumine (n = 23) or gadopentetate dimeglumine (n = 23) and then, after 2–7 days, a second examination with the other agent. Contrast administration (volume, rate), sequence parameters (T1wSE; T1wGRE), and interval between injection and image acquisition were identical for examinations in each patient. Three blinded neuroradiologists evaluated images qualitatively (lesion delineation, lesion enhancement, global preference) and quantitatively (lesion-to-brain ratio [LBR], contrast-to-noise ratio [CNR],%lesion enhancement). Differences were assessed using Wilcoxon’s signed-rank test. Reader agreement was determined using kappa (κ) statistics.
There were no demographic differences between groups. The three readers preferred gadobenate dimeglumine globally in 22 (53.7%), 21 (51.2%), and 27 (65.9%) patients, respectively, compared with 0, 1, and 0 patients for gadopentetate dimeglumine. Similar significant (P < 0.001) preference was expressed for lesion border delineation and enhancement. Reader agreement was consistently good (κ = 0.48–0.64). Significantly (P < 0.05) higher LBR (+43.5–61.2%), CNR (+51.3–147.6%), and % lesion enhancement (+45.9–49.5%) was noted with gadobenate dimeglumine.
Brain lesion depiction at 3T is significantly improved with 0.1 mmol/kg gadobenate dimeglumine.
brain tumor imaging; gadobenate dimeglumine; comparative studies; high field MRI
A 52-year-old woman presented with lower back pain, progressive symmetrical paraparesis with sensory impairment, and sphincter disturbance. Magnetic resonance imaging (MRI) of the whole spine revealed multiple intradural extramedullary serpiginous-mass lesions in the subarachnoid space continuously from the prepontine to the anterior part of the medulla oblongata levels, C7, T2-T8, and T12 vertebral levels distally until the end of the theca sac and filling-in the right S1 neural foramen. Sparganosis was diagnosed by demonstration of the sparganum in histopathological sections of surgically resected tissues and also by the presence of serum IgG antibodies by ELISA. DNA was extracted from unstained tissue sections, and a partial fragment of mitochondrial cytochrome c oxidase subunit 1 (cox1) gene was amplified using a primer set specific for Spirometra spp. cox1. After sequencing of the PCR-amplicon and alignment of the nucleotide sequence data, the causative agent was identified as the larva of Spirometra erinaceieuropaei.
Spirometra erinaceieuropaei; sparganosis; cauda equina syndrome; molecular identification
Recent advances in radiological and serological techniques have enabled easier preoperative diagnosis of sparganosis. However, due to scarcity of cases, sparganosis has been often regarded as a disease of other etiologic origin unless the parasite is confirmed in the lesion. We experienced a case of sparganosis mimicking a varicose vein in terms of clinical manifestations and radiological findings. Sparganosis should be included among the list of differential diagnosis with the varicose vein.
sparganosis; varicose vein; subcutaneous mass
Food-borne helminthiases (FBHs) have become increasingly important due to frequent occurrence and worldwide distribution. There is increasing demand for developing more sensitive, high-throughput techniques for the simultaneous detection of multiple parasitic diseases due to limitations in differential clinical diagnosis of FBHs with similar symptoms. These infections are difficult to diagnose correctly by conventional diagnostic approaches including serological approaches.
In this study, antigens obtained from 5 parasite species, namely Cysticercus cellulosae, Angiostrongylus cantonensis, Paragonimus westermani, Trichinella spiralis and Spirometra sp., were semi-purified after immunoblotting. Sera from 365 human cases of helminthiasis and 80 healthy individuals were assayed with semi-purified antigens by both a protein microarray and the enzyme-linked immunosorbent assay (ELISA). The sensitivity, specificity and simplicity of each test for the end-user were evaluated. The specificity of the tests ranged from 97.0% (95% confidence interval (CI): 95.3–98.7%) to 100.0% (95% CI: 100.0%) in the protein microarray and from 97.7% (95% CI: 96.2–99.2%) to 100.0% (95% CI: 100.0%) in ELISA. The sensitivity varied from 85.7% (95% CI: 75.1–96.3%) to 92.1% (95% CI: 83.5–100.0%) in the protein microarray, while the corresponding values for ELISA were 82.0% (95% CI: 71.4–92.6%) to 92.1% (95% CI: 83.5–100.0%). Furthermore, the Youden index spanned from 0.83 to 0.92 in the protein microarray and from 0.80 to 0.92 in ELISA. For each parasite, the Youden index from the protein microarray was often slightly higher than the one from ELISA even though the same antigen was used.
The protein microarray platform is a convenient, versatile, high-throughput method that can easily be adapted to massive FBH screening.
Food-borne helminthiases (FBHs) have caused significant problems in public health and also posed socio-economic concerns. Common FBHs, such as cysticercosis, trichinellosis, paragonimiasis, sparganosis and angiostrongyliasis, have a worldwide distribution with high morbidity and even death. The objective of the present study was to develop and test a rapid assay suitable for large-scale screening for FBHs that would also allow differential diagnosis between the various parasite species. We tested archived, well-characterized serum specimens and prioritized tests for future evaluation in rapid and simultaneous screening of five different FBHs, i.e. cysticercosis, trichinellosis, paragonimiasis, sparganosis and angiostrongyliasis. This was done with a multiplex protein microarray assay equipped with semi-purified antigens capable of detecting disease-specific antibodies. Results showed that the protein microarray developed displayed a good specificity, ranging from 97.0% to 100.0%, and a sensitivity, ranging from 85.7% to 92.1%, with a Youden index variation from 0.83 to 0.92. It was concluded that the protein microarray provides a sensitive, high-throughput technique for the simultaneous detection of multiple FBHs overcoming the limitations of conventional diagnostics.
A case of breast sparganosis was confirmed by surgical excision of a worm (fragmented into 5 pieces) in a 59-year-old Korean woman suffering from a palpable mass in the left breast. Mammography and ultrasonography characteristically revealed the presence of several well-defined, isodense and hypoechoic tubular masses, in the upper quadrant of the left breast, each mass consisting of a continuous cord- or worm-like structure. During surgery, a long segment of an actively moving sparganum of Spirometra sp. and 4 small fragments of the same worm, giving a total length of 20.3 cm, were extracted from the upper outer quadrant of the left breast and the axillary region. The infection source remains unclear, because the patient denied ingesting any snake or frog meat or drinking untreated water.
sparganum; sparganosis; case report; woman; breast; mammography; ultrasonography
The mature domain of a cysteine protease of Spirometra erinacei plerocercoid larva (i.e., sparganum) was expressed in Escherichia coli, and its value as an antigen for the serodiagnosis of sparganosis was investigated. The recombinant protein (rSepCp-1) has the molecular weight of 23.4 kDa, and strongly reacted with the sparganum positive human or mice sera but not with negative sera by immunoblotting. ELISA with rSepCp-1 protein or sparganum crude antigen (SeC) was evaluated for the serodiagnosis of sparganosis using patient's sera. The sensitivity and specificity of ELISA using rSepCp-1 protein were 95.0% (19/20) and 99.1% (111/112), respectively. In contrast, the sensitivity and specificity of ELISA with SeC were 100% (20/20) and 96.4% (108/112), respectively. Moreover, in experimentally infected mice, the sensitivity and specificity of both ELISA assays were 100% for the detection of anti-sparganum IgG. It is suggested that the rSepCp-1 protein-based ELISA could provide a highly sensitive and specific assay for the diagnosis of sparganosis.
Spirometra erinacei; sparganum; cysteine protease; ELISA; serodiagnosis
OBJECTIVE: To investigate the incidence and timing of neonatal ultrasound lesions, and clinical details about pregnancy and the perinatal period, in a total population of extremely premature children with cerebral palsy, born to mothers who were resident in Oxfordshire. METHODS: Eighteen children born at less than 32 completed weeks of gestation were identified from a regional cerebral palsy register. Eighteen controls were matched for gestation, time, and place of birth. Perinatal records and ultrasound reports were systematically reviewed. Sequential neonatal ultrasound images stored on videotape were reanalysed, blind to the outcome of the infants. RESULTS: Sixteen (89%) of the cerebral palsy cases and one (6%) control had parenchymal cysts on neonatal brain scans. Of the cerebral palsy cases, none had cysts detectable on the first day. Six developed cysts within the first 10 days of life, and two of these had periventricular echodensities when first scanned postnatally. Antenatal complications were recorded in 16 cases and nine controls. The early postnatal appearance of cysts in a few babies with a history of severe antenatal complications suggested that antenatal factors may have contributed to the cerebral pathology. CONCLUSIONS: Intrauterine factors may have contributed to adverse neurological outcome, but 16/18 of the preterm cerebral palsy cases had an associated cerebral lesion which developed in the perinatal period.
The present study reports a human case of cutaneous gnathostomiasis with recurrent migratory nodule and persistent eosinophilia in China. A 52-year-old woman from Henan Province, central China, presented with recurrent migratory reddish swelling and subcutaneous nodule in the left upper arm and on the back for 3 months. Blood examination showed eosinophila (21.2%), and anti-sparganum antibodies were positive. Skin biopsy of the lesion and histopathological examinations revealed dermal infiltrates of eosinophils but did not show any parasites. Thus, the patient was first diagnosed as sparganosis; however, new migratory swellings occurred after treatment with praziquantel for 3 days. On further inquiring, she recalled having eaten undercooked eels and specific antibodies to the larvae of Gnathostoma spinigerum were detected. The patient was definitely diagnosed as cutaneous gnathostomiasis caused by Gnathostoma sp. and treated with albendazole (1,000 mg/day) for 15 days, and the subsequent papule and blister developed after the treatment. After 1 month, laboratory findings indicated a reduced eosinophil count (3.3%). At her final follow-up 18 months later, the patient had no further symptoms and anti-Gnathostoma antibodies became negative. Conclusively, the present study is the first report on a human case of cutaneous gnathostomiasis in Henan Province, China, based on the past history (eating undercooked eels), clinical manifestations (migratory subcutaneous nodule and persistent eosinophilia), and a serological finding (positive for specific anti-Gnathostoma antibodies).
Gnathostoma spinigerum; gnathostomiasis; migratory subcutaneous nodule; serodiagnosis; albendazole; China
To describe a previously unreported clinical entity superficially resembling macular serpiginous choroiditis but with a distinct presentation and clinical course.
A retrospective review of the medical records of five patients, aged 50 to 68 years, exhibiting this entity seen at five different centers from 1999 to 2006.
The lesions in the patients in this study are in some respects similar to those of acute macular serpiginous choroiditis. The patients had well-delineated whitish plaque-like lesions involving the macula and sparing the peripapillary areas of both eyes. In contrast to serpiginous choroiditis, visual acuity remained good despite early involvement of the fovea until complications related to choroidal neovascularization (CNV) or pigmentary mottling developed. The angiographic characteristics and the clinical course were also atypical. Fluorescein angiography revealed well-defined early hypofluorescent areas, which partially filled-in in the late phase. Indocyanine green angiography showed the hypofluorescence to be persistent. Unlike serpiginous choroiditis, the white macular lesions faded over a period of months to years, but the characteristic angiographic findings often persisted longer. CNV developed in nine of 10 eyes with subsequent conversion to disciform macular scars in seven of 10 eyes. Unlike serpiginous choroiditis, none of the eyes showed chorioretinal scar formation unless related to CNV.
Persistent placoid maculopathy has features resembling macular serpiginous choroiditis but differs in its clinical course and effect on visual acuity. It appears to be a new entity. The majority of eyes develop CNV, which results in loss of central vision.