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EUR J PHYS REHABIL MED 2015;51:337-9

Factitious lymphedema of the arm: case report and review of publications

The aim of this study was to report a case of factitious lymphedema of the arm and related lymphoscintigraphic aspects. The case of a 36-year-old patient is reported who started to present with pain, in the 3rd finger of the right hand three years prior to this report, which she associated with her work. Joint effusion was identified and treated using a splint that restricted blood flow leading to edema of the distal third of the forearm. Since then the patient was treated however her condition worsened resulting in edema of the entire arm. Subsequently she was referred to our service. A physical examination identified a restrictive band in the axillary region of the arm that delimited the edema. Volumetry and lymphoscintigraphic examinations of the limb were performed. The lymphoscintigraphy demonstrated acceleration of the flow in the affected limb and dermal reflux. Clinical treatment with removal of the restriction allowed a rapid reduction in the volume of the limb. Key words: Lymphedema - Factitious disorders - Therapeutics.

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ymphedema is characterized as a type of edema due to an abnormal accumulation of fluids and other substances in the tissues resulting from a failure of the lymph drainage system.1, 2 Factitious lymphedema is a deliberate form of the disease in which the patient repetitively or for a long period of time applies a tourniquet around the limb thereby causing edema.2-9 This condition was initially called Correspondence author: J. M. Pereira de Godoy, Rua Floriano Peixoto 2950, São José do Rio Preto-SP, CEP:15020-010. E-mail: [email protected]

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IN C ER O V P A Y R M IG E H DI T C ® A

M. DE FÁTIMA GUERREIRO GODOY 1, J. M. PEREIRA DE GODOY

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CASE REPORTS

1Medicine School, São José do Rio Preto FAMERP and Research Godoy Clinic, Brazil 2Department of the Cardiology and Cardiovascular Surgery in Medicine School of São José do Rio Preto- FAMERP and Research CNPq (National Council for Research and Development). Brazil.

Secretan’s disease in a patient with trauma and edema of the dorsal metacarpus.3 This disease has been associated with psychiatric disorders.2, 10, 11 Pain may occur from self-mutilation-associated with anatomic dysfunction, edema, and reduction of movements and consequently atrophy.12 Histological examinations demonstrate that at the site of restriction there is a lesion of the vessels due to the restriction and a dilation probably affecting lymphatic collectors. This disease has a classification similar to other forms of secondary lymphedema.11 The aim of this study was to report a case of factitious lymphedema of the arm and to describe lymphoscintigraphic characteristics. Case report The case of a 36-year-old patient is reported who started to present with pain in the 3rd finger of the right hand three years prior to this report which she associated with her work. Joint effusion was identified and treated using a splint that restricted blood flow leading to edema of the distal third of the forearm (Figures 1A, B, 2). Since then

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FACTITIOUS LYMPHEDEMA OF THE ARM

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Figure 1.—A, B) Edema of the distal third of the forearm.

Figure 2.—Lymphoscintigraphic study showing acceleration of the flow in the affected limb.

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DE FÁTIMA GUERREIRO GODOY

the patient was treated however her condition worsened resulting in edema of the entire arm. Subsequently she was referred to our service. A physical examination identified a restrictive band in the axillary region of the arm that delimited the edema. The patient alleged that she started to use a bandage to hide a small blemish in that region. Volumetry and lymphoscintigraphy of the limb were performed. The lymphoscintigraphic study demonstrated acceleration of the flow in the affected limb (Figure 2) but a film demonstrated dermal reflux (Figure 3). Treatment included identifying psychological aspects, removal of the tourniquet and lymph drainage. The study received the approval of the Local

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Figure 3.—Film showing dermal reflux.

Ethics Research Committee (Medicine School of São Jose do Rio Preto -approval n0 0573/2008) and participants signed written consent forms.


June 2015

DE FÁTIMA GUERREIRO GODOY

References

The current study shows a case of factitious lymphedema and demonstrates the lymphoscintigraphic pattern in these situations. However, there are only 13 citations of factitious lymphedema reported in PubMed with the majority involving the hands and only one involving the arm. In the current study, edema was initially limited to the distal third of the forearm and hand but later affected the entire arm. Use of a splint triggered a flow restriction but after the patient restricted the flow in the axillary region. An interesting aspect of this case is the lymphoscintigraphic finding due to the small number of published cases. A hyperdynamic pattern of drainage was observed in the affected limb after removing the restriction. However the lymphoscintigraphic film shows dermal reflux, which associated with the clinical findings, confirmed the diagnosis of factitious lymphedema. One published study on this issue did not show any reduction in the transport of lymph,14 but another study reported acceleration of the transport of lymph.15 Initially constriction using a tourniquet results in a venous return flow disorder and a rapid decompensation of the lymphatic return due to an increase in the transport of lymph. Mechanical insufficiency affects the lymphatic system depending on the force and duration of the pressure exerted by the tourniquet. The result is a complication typical of chronic lymphostasis. Many theories have been suggested for the disease although there does not seem to be a consensus on the physiology, classification, prognosis or treatment as there is psychological involvement affecting the entire physical and social dynamics of these patients.

1. Depairon M, Pittet L, Paillex R, Klumbach D, Mazzolai L. To live with lymphedema: present and future. Rev Med Suisse 2009;5:299-302. 2. de Godoy JM, de Godoy Mde F. Godoy & Godoy technique in the treatment of lymphedema for under-privileged populations. Int J Med Sci 2010;7:68-71. 3. Mortimer P. Differential diagnosis of chronic swelling of the limbs. I: Browse NS, Burnand KG, Mortimer PS, editors. Disease of the lymphatics. London: Arnold Publishers; 2003. p. 158-66. 4. Moretta DN, Cooley RD Jr. Secretan’s disease: a unique case report and literature review. Am J Orthop 2002;31:524-7. 5. Rabe E. Artificial lymphedema from the clinical view. Wien Med Wochenschr 1999;149:95. 6. Stoberl C, Musalek M, Partsch H. Artificial edema of the extremity. Hautarzt 1994;45:149-53. 7. Orenstein A, Friedman B, Yaffe B, Blankstein A, Tsur H. Factitious lymphedema of the hand: a diagnostic challenge. Cutis 1987;39:427-8. 8. Chatterjee T, Frey M, Zehnder T. Unusual lymphedema of the upper extremity. Schweiz Med Wochenschr 1999;129:292. 9. Godoy JMP, Godoy MFG, Spiandorin D, Valente FM. Factitious lymphedema: case report and literature review. J Vasc Br 2005;4:98-100. 10. de Fontaine S, Van Geertruyden J, Preud’homme X, Goldschmidt D. Munchausen syndrome. Ann Plast Surg 2001;46:153-8. 11. Louis DS, Lamp MK, Greene TL. The upper extremity and psychiatric illness. J Hand Surg 1985;10:687-93. 12. Schwartzman RJ. New treatments to reflex sympathetic dystrophy. N Engl J M 2000; 343:654-6. 13. Stöberl C, Musalek M, Partsch H. Artificial edema of the extremity. Hautarzt 1994;45:149-53. 14. Schuchhardt CH, Weissleder H. Artificial lymphedema. In: Schuchhardt CH, Weissleder H, editors. Lymphedema diagnosis and therapy. Germany: Viavital Verlag; 2001. p. 175-86. 15. Kittner C, Kroger J, Rohrbeck R, Parnitzke B, Decker S, Lakner V. Lymphatic outflow scintigraphy in a case of artificial oedema of the lower limb. Nuklearmedizin 1994;33:268-70.


Discussion

Conflicts of interest.—The authors certify that there is no conflict of interest with any financial organization regarding the material discussed in the manuscript. Received on July 28, 2012. Accepted for publication on February 16, 2015. Epub ahead of print on February 18, 2015.

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