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Isolated penile and scrotal elephantiasis presents a diagnostic and therapeutic challenge. We report a case of chronic penile and scrotal elephantiasis caused by indolent Chlamydia trachomatis infection. The patient improved modestly with long-term doxycycline.
Elephantiasis nostras is a rare condition that usually presents as a persistent swelling of the lower extremity secondary to recurrent lymphangitis. We present a patient, originally referred for "angioneurotic edema of the lip," who presented with a history of several months of persistent swelling of the upper lip. There was probable evidence for recent infection of the lip that is consistent with the diagnosis of elephantiasis nostras. This disease should be included in the differential diagnosis of chronic persistent angioedema of the lip.
A recent case-control study in Indonesia suggested that the course of Brugian filariasis and in particular resistance to the development of elephantiasis was associated with certain HLA class II alleles. In order to see whether these data could be confirmed we conducted a similar study in another Indonesian population from South Sulawesi. We could not confirm our earlier results and therefore concluded that HLA-DR and -DQ alleles are at least not strongly associated with progression to elephantiasis in Brugian filariasis. The complete data are presented also for anthropological reference purposes.
The case of a newborn girl with a rare, giant, congenital, tissue lymphangioma giving rise to elephantiasis of the right lower limb is presented. The different imaging methods, especially magnetic resonance imaging, showed no extension of the lesions into the deep structures. At the age of 2 years, the child underwent a roentgenographic skeletal survey, which revealed osteolytic lesions in the femurs and the right tibia. There was no clinical evidence of systemic involvement. The place of this affection among the different lymphatic malformations was discussed and the diagnosis of elephantiasis-like lymphangiomatosis of the limb, an extremely rare disorder, has been retained. Early surgical reduction was performed, followed by application of a pressure dressing. Five years later the result remains satisfactory, but the excision of a persistent fluid-filled pouch around the knee will probably be necessary in the future.
Chronic filaritic lymphoedema and elephantiasis, in India, were treated orally with 5,6 benzo-alpha-pyrone (56 BaP; 1,2 benzo-alpha-pyrone; coumarin) in a double-blind, randomized, matched-group trial. Each group finally contained 40-55 patients. Patients were observed for about 2 years (ranging from 6 to 45 months, with 75% completing the 2 years). The 56 BaP slowly, but very significantly (P < 0.0001), reduced all grades of lymphoedema and elephantiasis. Two thirds of the oedema was lost by grade 2 over 2 years. Grades 3 to 5 were reduced by a fifty over that time. The greater the initial oedema, the greater was its rate of resolution. A slowly worsening condition thus became a slowly improving one. Slowness has its advantages: compression stockings, that are impractical in hot, wet or dirty conditions, are not necessary. The slowly remodelling fibrous tissue, while lessening in amount, is still able to hold the tissues together. The 56 BaP considerably improved many symptoms and complications, particularly bursting-pains, inflammation and ulcers. It is cheap and of very low toxicity. Diethylcarbamazine (DEC) was studied with and without 56 BaP. DEC alone gave some reduction of the oedema, but this was much smaller than that with 56 BaP. It considerably worsened the reductions by 56 BaP, while 56 Bap slightly improved those by DEC and reduced the fever caused by DEC. Together, they reduced feelings of swelling and bursting-pain, fungal infections, lymphangitis and lymphadenitis more than when used alone.
From 1988 to 2005, 8 men who presented with penoscrotal elephantiasis underwent penile shaft degloving and reduction scrotoplasty, followed by transplantation of a split-thickness skin graft (STSG) to the penile shaft. The etiology of elephantiasis in these patients included self-injection of viscous fluid and postsurgical obstructive lymphedema. In the 6 most recent cases, negative-pressure dressings were applied over the STSG to promote graft take, and STSG take rate was 100%. The results of our series corroborate those of a previous report, which showed circumferential negative-pressure dressings to be safe and efficacious in bolstering STSGs to the penile shaft. Furthermore, these results suggest that the use of negative-pressure dressings may improve graft take in this patient population.
Genital elephantiasis (esthiomene), which is the dramatic end-result of lymphatic obstruction, is rather rare. Although mainly associated with filariasis and sexually transmitted diseases, such as lymphogranuloma venereum and donovanosis, it could also be an uncommon complication of tubercular lymphadenitis, a common infection in tropical countries. We report a rare case of a 32-year-old Indian female in whom genital elephantiasis occurred as a complication of tubercular lymphadenitis.
Angioedema of the face is a common entity seen by the allergist, but other less common conditions should also be considered in the differential diagnosis of facial swelling. Elephantiasis nostras is a rare condition that can result in persistent swelling of the lips secondary to recurrent attacks of lymphangitis caused by bacterial infection. Cheilitis granulomatosa is a disorder that may also present with lip swelling. We present a patient with persistent swelling of his lips who was referred to our service to consider an allergic cause. The clinical findings of persistence of the swelling without improvement after therapy with corticosteroids and antihistamines are considered consistent with elephantiasis nostras or cheilitis granulomatosa. The histopathologic findings in this case revealed epithelioid cell granulomas, which are consistent with cheilitis granulomatosa. These two disorders should be included in the differential diagnosis of localized swelling of the face.
Two cases of buphthalmos and regional gigantism in peripheral neurofibromatosis (Von Recklinghausen disease) are described. The ocular hypertension was detected at birth and thereafter the facial hemihypertrophy, café-au-lait spots and neurofibromas. One case presented a progressive facial elephantiasis without buphthalmos. The evolution in neurofibromatous elephantiasis of these three cases is discussed.
The distribution of non-filarial elephantiasis of the lower legs in Rwanda and Burundi in Central Africa has been studied in order to test the previous observation of an association between the disease and the volcanic rocks and soils on which the people live. Using the method of market counts, a total of 26,602 adults were observed in 23 markets; 189 had elephantiasis. In addition, 264 cases were observed in specially arranged clinics and a further 77 while travelling. Prevalence per thousand adults varied from 0.0 to 20.7. It was noted that the areas of high prevalence corresponded with the areas of volcanic lava. A differential market count at the edge of the red soils in two of these areas showed a raised prevalence among people living on these soils, similar to that observed in Ethiopia. The observations confirm that the disease occurs in barefooted people who dig in soil developed from volcanic rocks, under tropical conditions, at altitudes around 1500 m. It is suggested that the high proportion of iron and other transitional metals may be important as irritant or toxic to the lymphatic vessels of the legs after absorption.
The most characteristic lesions of neurofibromatosis are the extremely large plexiform neurofibromas involving an entire extremity, which give rise to the condition known as elephantiasis neuromatosa. In this article, we present a patient who was diagnosed as elephantiasis neuromatosa with Becker's melanosis clinically and review the literature briefly.
Penoscrotal elephantiasis is a rare disease outside of filariasis endemic regions. The authors report a case of primary penoscrotal elephantiasis treated by complete surgical resection of the pathological tissue and penoscrotal reconstruction, with a good functional and cosmetic result. The complementary investigations indicated and the various treatment options are analysed in the light of a review of the literature.
Scrotal elephantiasis (scrotal lymphoedema) is essentially observed in filaria endemic zones. It is very rare outside of these zones and is usually idiopathic, and rarely congenital or secondary. The authors report two cases of scrotal elephantiasis treated by wide resection of the pathological scrotal wall and penoscrotal repair with a good functional and cosmetic result.
Elephantiasis nostras verrucosa represents an uncommon yet distinct clinical entity resulting from chronic lymphedema of an extremity or body region. Characterized by profound non-pitting edema with cobblestone-like papules, plaques, and nodules, it typically occurs secondary to infections, surgeries, tumor obstruction, radiation, congestive heart failure, and obesity. This progressively deformative disorder has been treated with various medical and surgical measures. In the following case report, the history, clinical, and pathologic appearance of elephantiasis nostras verrucosa are discussed, as well as newer treatment options.
Elephantiasis nostras is characterized by edema, skin fibrosis, and massive enlargement of a body part. Lymphatic obstruction, most commonly due to surgery, radiation, infection, or neoplasms, is important in its pathogenesis. The diagnosis of elephantiasis nostras can often be made based on the clinical findings, but examination of tissue may be helpful to rule out associated conditions, especially malignancies. Mainstays of therapy are elevation, use of pressure devices, and administration of antibiotics. Although medical and surgical treatments are limited in their value, pneumatic pumps are effective in refractory cases.
Elephantiasis Nostras Verrucosa (ENV) is a rare form of pretibial myxedema, which is nearly always associated with Graves' disease. A case is presented here of Graves' disease who had elephantiasis variety of pre-tibial myxedema (PTM).
Scrotoplasty has been used in the treatment of scrotal elephantiasis but it is most traumatic and achieved unsatisfactory results. The author developed a microlymphatico-venous procedure to treat elephantiasis of the scrotum and applied it clinically with good results. The scrotal size was immediately reduced to a nearly normal value. The indication, types of procedures and advantages of the operation are presented.
Twenty-five years after the fight started against Wuchereria bancrofti var. pacifica in French Polynesia and which was based upon mass chimiotherapy by diethylcarbamazine (Banocide), a survey realized in 1975 and 1976 among 274 patients with elephantiasis allows us to give precise details of the local clinical and epidemiological aspects. Since 1950 the number of clinical cases varies along with the parasitological incidence, but with a certain delay which is explained by the late and irreversible elephantiasis characteristics. The overall prevalence is actually low. However the occurrence of new cases, although very rare, is still possible. Among sick patients the lymphangitic crisis frequency whose aspect lead to evoke a bacterial participation, is low. Women are less frequently and less severely involved than men. Localisations are essentially restricted to the lower limbs. Several limbs are involved in approximately 50 0/0 of the cases. The scrotum involvement is rare, the one of the breast is found only once. Among men we can notice a 33 0/0 incidence of associated hydroceles. A past history of chyluria is not rare.