Lymphedema as a presenting sign of toxocariasis

Case Report J. A m i r , L. H a r e l , T. E i d l i t z - M a r k u s , I. V a r s a n o

Lymphedema as a Presenting Sign of Toxocariasis Summary: Toxocariasis in children is usually an asymptomatic infection and those with clinical illness have non-specific systemic or local manifestations. We present a 24-month-old boy with bilateral lymphedema of the feet as the main clinical manifestation of toxocariasis. The child presented with limping and nonpitting edema of both feet. Laboratory investigation revealed leucocytosis of < 20,000/mm 3 with a differential count of < 50% eosinophils. No other cause of edema was found. The ELISA for toxocariasis revealed a high titer of >- 1:4,096. The limping and the lymphedema disappeared during the third week of his illness. We suggest that toxocariasis should be considered as a possible cause of lymphedema and eosinophilia in young children. Introduction Toxocariasis is mainly caused by Toxocara canis and is a common zoonosis of children wherever dogs are found. Most seropositive children are asymptomatic [1] and those with clinical illness usually have non-specific systemic or local manifestations [2, 3]. The diagnosis of toxocariasis is usually considered in the differential diagnosis of leucocytosis with marked eosinophilia (of more than 30%) or in the investigation of inflammatory mass in the eye. We present a young child with bilateral lymphedema of the feet as the main clinical manifestation of toxocariasis.

Case Report A 24-month-old boy was seen in the pediatric emergency room of Hasharon Hospital with a 2-day history of fever and limping on his right leg. SweUing of the right foot was treated as cellulitis with cephalexin. On the second visit, 4 days later, his gait had improved but upon physical examination, prominent nonpitting edema of both feet was noticed. The skin of the foot was not red or warm and the ankle joints had a full range of motion. Hepatomegaly was the only other positive physical finding. On followup visits, fever between 37.3-38.8°C was measured during the first 7 days, and the lymphedema of the feet gradually disappeared during the third week of his illness. An ultrasonographic scan of the abdomen and pelvis revealed only homogenous liver enlargement, and the chest x-ray was unremarkable. Laboratory investigation revealed WBC in the range of 21,000-25,000/mm 3 with a differential count of 51-57% eosinophils, 8-16% segments, 23-30% lymphocytes and 2-5% monocytes. Hemoglobin was 9.8g/dl with mild hypochromia and microcytosis, thrombocytosis of 619,000 mm3, ESR 80ram/h, aspartate aminotransferase activity (AST, SGOT) 215 U/l, alanine aminotransferase (ALT, SGPT) 245, gamma glutamyl transferase 11 U/I, creatinine phosphokinase 71 mg/dl, albumin 4.1 g/dl, globulin 3.7g/dl, urea 27mg/dl, creatinine 0.55 mg/dl, IgG 1550 mg/dl, IgA 59 mg/dl, IgM 180 mg/dl. Several stool specimens for parasites revealed cysts of Giardia lamblia and Entamoeba histolytica. The enzyme-linked immunosorbent assay for toxocariasis (kindly performed at the Centers of Disease Control in Atlanta, GA), revealed a very high positive titer of -> 1:4,096 (positive test --- 1:32). Repeat laboratory studies, 3 weeks after his initial examination, revealed normal liver enzymes but the leucocytosis and eosino-

philia were still present at the time of submission of this paper

(6 months later).

Discussion The clinical manifestation of toxocariasis was traditionally divided into two principal forms: visceral larva migrans and ocular toxocariasis (OT). Recently, a third clinical form called ,,covert toxocariasis" has described patients with positive serology and various symptoms and signs. These include gastrointestinal disorders (notably abdominal pain), behavorial disturbances, weakness and lethargy [4-6]. In a large number of children with visceral larva migrans Huntly et al. [2] found that cough, wheezing, fever and convulsions were the main symptoms. The physical findings included hepatomegaly, tales, wheezing, skin lesions and lymphadenopathy. The child presented here had visceral larva migrans diagnosed by leucocytosis with marked eosinophilia and high titer of antibodies to toxocara. There was no history of pica, but the presence of at least two other kinds of parasites in the stool suggested ingestion of dirt [2]. The prominent lymphedema which caused the limp lasted over 2 weeks. Limb pain [4], edema of the calves [7] and transient myositis [8] were described in the past as associated with toxocariasis. Treatment of toxocariasis is controversial. Although most cases recover without specific therapy, as did our patient, antihelminthic drugs such as thiabendazole may be considered in severe cases. Lymphedema of the foot is secondary to obstruction of the regional lymphatic drainage. We speculate that the inflammatory process in the regional lymph nodes due to parasite migration was probably the cause of the lymphedema in the presented child, even though no lymphadenopathy was noticed on physical examination. A very striking

Received: 2 February 1995/Revision accepted: 7 July 1995 J. Amir, M. D., Liora Harel, M. D., Tal Eidlitz-Markus, M. D., L Varsano, M. D., Dept. of Pediatrics "C", Children's Medical Center in Israel, Sackler School of Medicine, Tel Aviv University, P. O. Box 559, Petach Tikvah, 49202 Israel.

Infection 23 (1995) No.6 © MMV Medizin Verlag GmbH Mtinchen, Miinchen 1995

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J. A m i r et al.: Lymphedema as Presentation of Toxocariasis cause of chronic inflammatory obstruction of the lymphatics is e n c o u n t e r e d in t h e p a r a s i t i c i n f e c t i o n of Wuchereria bancrofti w h i c h causes e l e p h a n t i a s i s . H o w e v e r , in t o x o c a riasis t h e p r o c e s s is transient.

Zusammenfassung: Lymphiidem als Primiirsymptom einer Toxocariasis. Die Toxocariasis nimmt bei Kindern in der Regel einen asymptomatischen Verlauf. Wenn Krankheitszeichen auftreten, handelt es sich um unspezifische AUgemeinsymptome oder lokale Symptome. Wir berichten tiber einen 24 Monate alten Jungen, bei dem ein bilaterales Lymph6dem der FtiBe die Hauptmanifestation der Toxocariasis war. Das Kind hinkte und hatte an beiden Fiigen ein nicht dellenbildendes (3dem.

W e suggest t h a t t o x o c a r i a s i s s h o u l d b e c o n s i d e r e d as a p o s s i b l e c a u s e of l y m p h e d e m a a n d e o s i n o p h i l i a in y o u n g children.

Dabei bestand eine Leukozytose v0n > 20 000/ram 3 mit > 50% Eosinophilen im Differentialblutbild. Andere Ursachen ftir das ()dem waren nicht zu finden. Der E L I S A ftir Toxocariasis ergab einen hohen Titer von > 1:4 096. Hinken und Lymph6dem verschwanden im Verlauf von drei Wochen. Wir verweisen auf die M6glichkeit einer Toxocariasis als m6gliche Ursaehe for Lymph6dem und Eosinophilie bei kleinen Kindern.

References 1. Jones, W. E., Schantz, P. M, Foreman, K., Smith, L K., Witte, E. J., Schooley, D. E., Juraneck, P. D.: Human toxocariasis in a rural community. Am. J. Dis. Child t34 (1980) 967-969. 2. Huntley, C. C., Costas, M. C., Lyedy, A.: Visceral larva migrans syndrome: clinical characteristics and immunologic studies in 51 patients. Pediatrics 36 (1965) 523-536. 3. Worley, G., Green, J. A., Frothingham, T. E., Sturner, R. A., Walls, K. W., Pakalnis, V. A., Ellis, G. S.: Toxocara canis infection: clinical and epidemiologicalassociations with seropositivityin kindergarten children. J. Infect. Dis. 159 (1984) 591-596. 4. Taylor, M. R. H., Keane, C. J., O'Connor, P., Mulvihill, E., Holland, T.: The expanded spectrum of toxocaral disease. Lancet i (1988) 692-694.

5. Rasmussen, L. N., Dirdal, M., Birkebeck, N. H.: ,,Covert toxocariasis" in a child treated with low-dose diethylcarbamazine. Acta Paediatrica 82 (1993) 116-118. 6. Natwani, D., Loing, R. B, Currie, P. E.: Covert toxocariasis, a cause of recurrent abdominal pain in childhood. Br. J. Clin. Pract. 46 (1992) 271-273. 7. Ben-Ami, M., Katzuni, E., Hochman, H., Antonelli, J., Koren, A.: Toxocariasis in Emek Israel. Harefuah (Hebrew) 119 (1990) 72-73. 8. Walsh, S. S,, Robson, W. J., Hart, C. A.: Acute transient myositis due to toxocara. Arch. Dis. Child 63 (1988) 1087-1088.

Book Review M. Grossmann, J. Roth Cutaneous Manifestations of Infection in the Immunocompromised Host 196 pages, numerous figures Williams & Wilkins, Baltimore 1995 Price: $ 32.95 There are certain topics which regularly attract potential authors. This book constitutes a prime example. In this case, however, this is to the advantage of the reader. H e has the author to thank for a brilliant text book giving concise statements based on topical references and a large series of impressive clinical photographs. Unfortunately, the quality of reproduction of the colour photographs is not optimal. This could be due only in part to the particular circumstances under which the photograph had to be taken. One would certainly not like to subscribe to every single statement, e. g., I would rather agree with the statement on p. 62: "dermatophytes typically do not invade living tissue" than subscribe to the interpretation of the lesions demonstrated on p. 66

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(Fig. 3.8) as "papulonodules of invasive T. rubrum infection". In the first place, as far as can be judged from the photograph there are no true nodules but rather reddish macules, papules and pustules. Moreover, the distribution is follicular as far as papules and pustules are concerned. Thus it is follicular fungal rather than true invasive disease. While the major part of the b o o k consists of special articles on the various diseases to be dealt with, there is a short introductory and a somewhat longer concluding section giving very helpful additional information. This in particular applies to the table on differential diagnoses oriented at various clinical pictures, even giving topical references. Finally, it should be lauded that, unlike that of most corresponding books, the focus here is not merely on cutaneous infection in H I V disease. It can be therefoi"e recommended to every doctor in charge of the care of patients who are critically ill due to a compromised immune system.

H. C. Korting Mtinchen

Infection 23 (1995) No. 6 © MMV Medizin Verlag GmbH Mtinchen, Mt~nchen 1995