Combined Conservative Treatment and Lymphatic Venous Anastomosis for Severe Lower Limb Lymphedema with Recurrent Cellulitis

Accepted Manuscript Combined conservative treatment and Lymphatic Venous anastomosis for severe lower limb lymphedema with recurrent cellulitis Makoto Mihara, M.D., Hisako Hara, M.D., Hiromi Tsubaki, NS, Takiko Suzuki, DT, Naomi Yamada, DT, Mari Kawahara, M.D., Noriyuki Murai, M.D. PII:

S0890-5096(15)00399-4

DOI:

10.1016/j.avsg.2015.01.037

Reference:

AVSG 2383

To appear in:

Annals of Vascular Surgery

Received Date: 9 December 2014 Accepted Date: 28 January 2015

Please cite this article as: Mihara M, Hara H, Tsubaki H, Suzuki T, Yamada N, Kawahara M, Murai N, Combined conservative treatment and Lymphatic Venous anastomosis for severe lower limb lymphedema with recurrent cellulitis, Annals of Vascular Surgery (2015), doi: 10.1016/ j.avsg.2015.01.037. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

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Combined conservative treatment and Lymphatic Venous anastomosis for severe lower limb lymphedema with recurrent cellulitis

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Makoto Miharaa*, M.D., Hisako Haraa,b*, M.D., Hiromi Tsubaki NSc, Takiko Suzuki DTd, Naomi Yamada DTd, Mari Kawahara M.D.a, Noriyuki Murai M.D.a Institutions: Department of Vascular Surgery, Saiseikai Kawaguchi General Hospital, Saitama, Japan b Department of Plastic and Reconstructive Surgery, The University of Tokyo Hospital, Tokyo, Japan c Department of Nursing, Saiseikai Kawaguchi General Hospital, Saitama, Japan d Department of Nutrition, Saiseikai Kawaguchi General Hospital, Saitama, Japan

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* Equal Contribution

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RUNNING HEAD: LVA and conservative treatment for severe lymphedema CORRESPONDENCE TO:

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Makoto Mihara M.D.

Department of Vascular Surgery, Saiseikai Kawaguchi General Hospital, Saitama, Japan

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332-0021 Nishi-Kawaguchi 5-11-5, Kawaguchi-Shi, Saitama, Japan E-mail: [email protected]

Tel and Fax; 048-253-1551

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Abstract Background: Lymphedema may be treated either conservatively or surgically. Although conservative therapy is the first-line treatment, some patients are refractory

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to it and repeat severe cellulitis. We usually perform lymphatico-venous anastomosis (LVA) for lymphedema patients, and LVA can reduce the frequency of cellulitis.

Case report: A 67-year-old woman. She had undergone a radical hysterectomy, pelvic lymphadenectomy, and postoperative radiotherapy, for cervical cancer at the age 50 years. She developed lymphedema in both legs and high-pressure compression

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stockings caused lymphorrhea in the groin and thigh, resulting in recurrent episodes of cellulitis. Lymphoscintigraphy revealed dilation of the lymphatic vessels in both legs. Results of an indocyanine green test revealed dermal backflow throughout the lower

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body. After wearing low-pressure stocking, we performed LVA to reduce cellulitis. After confirming the result of LVA the patients started wearing high-pressure stocking. The patient underwent a subsequent LVA, 3 months after the first, to further improve edema. The lymphorrhea resolved, and cellulitis did not recur.

Conclusions: The combination of surgical treatment and conservative treatment is important for severe lymphedema treatment. Although conservative treatment is usually said to be the first-line, LVA can antecede in cases refractory to conservative

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treatment.

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Abbreviations:

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LVA; lymphatic venous anastomosis

QOL; quality of life

ICG; indocyanine green

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1. Introduction

Lymphedema may be treated either conservatively1 or surgically.2,3 Although conservative therapy is the first-line treatment, some patients develop cellulitis under a conservative regimen. In such cases, it may be necessary to discontinue conservative

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therapy, and improvement in lymphedema is difficult to achieve.4 Without effective therapy, symptoms exacerbated. While conservative therapy is known to reliably reduce the frequency of cellulitis, for patients who cannot be treated with conservative therapy, an alternative method is required. Administration of antibiotics has been found to inhibit the development of cellulitis.5

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Recent reports have indicated that lymphatic venous anastomosis (LVA) may reduce the incidence of cellulitis;6 this surgical approach may be an option for patients who cannot undergo treatment with conservative therapy.

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Here, we report the case of a patient with recurrent cellulitis that was successfully controlled with a combination of conservative and surgical therapy. 2. Patient

A 67-year-old woman presented with recurrent cellulitis; the patient had undergone a radical hysterectomy, pelvic lymphadenectomy, and postoperative radiotherapy (50 Gy), for cervical cancer at the age 50 years. Immediately after radiotherapy, she developed

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lymphedema in both legs (Figure 1). She presented to a clinic that administered conservative therapy, and started treatment with elastic compression stockings. However, the high-pressure compression stockings caused lymphorrhea in the groin and thighs, resulting in recurrent episodes of cellulitis. These episodes of cellulitis

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necessitated discontinuation of conservative therapy, which in turn worsened lymphedema; thus, the patient’s symptoms and lymphedema deteriorated in a vicious

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cycle. Cellulitis recurred with a frequency of approximately 4 times a year. The patient became depressed and suicidal. After cancer treatment, she became confined to the house and her quality of life (QOL) markedly deteriorated; hence, she was brought to our department by family members. Lymphoscintigraphy revealed dilation of the lymphatic vessels in both legs. The inguinal lymph nodes were visualized after 15 min, suggesting that lymphatic vessel function was well preserved (Figure 2). Results of an indocyanine green (ICG) test revealed dermal backflow throughout the lower body (Figure 3). We considered surgical therapy, but initially attempted to improve the lymphedema as far as possible with low-pressure compression stockings (10 mmHg). Two months after the patient started wearing the stockings, however, she again developed cellulitis.

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Therefore, control of lymphedema with conservative therapy alone was not possible, and LVA was performed under local anesthesia. During the operation, an ectatic lymph node, dilated to around twice its normal size (diameter, 0.8 mm) was observed (Figure 4). There was minimal sclerosis of the lymphatic vessels. The operating time was 3 h 30

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min, with 2 anastomoses performed in the right leg, and 4 in the left (Figure 5a). The patient changed to moderate-pressure compression stockings (20 mmHg) at 1 month postoperatively. The lymphorrhea resolved, and cellulitis did not recur. The patient underwent a subsequent LVA, 3 months after the first, to further improve edema (Figure 5b). The operating time was 3 h 57 min, with 4 anastomoses performed in the

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right leg, and 3 in the left. After it had been confirmed that the cellulitis had not recurred, the patient switched to high-pressure compression stockings (30 mmHg). Lymphorrhea and cellulitis did not recur, and the circumference of the patient's legs

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considerably improved (Figure 6). Because of the resolution of the cellulitis and improvement in the edema, the patient’s QOL improved, and she started to attend a local weekly exercise class.

3. Discussion

We combined conservative therapy1,2 and surgical therapy3,4 to successfully treat a

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patient with recurrent cellulitis and associated aggravated lymphedema. Currently, standard conservative therapy for lymphedema consists of complex decongestive therapy, which includes the use of high-pressure compression stockings or bandages, and manual lymph drainage. However, a significant number of patients drop

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out of therapy because of the physical and mental stress involved.7 Patients may have difficulty wearing high-pressure compression stockings, either because of psychological

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or physical conditions such as rheumatoid arthritis or old age. In such patients, lymphedema must be controlled with the use of moderate-pressure or low-pressure stockings; combining the use of compression with surgical therapy ,especially lymphatic venous anastomosis, may be helpful in such cases. As in the present case, the use of surgery may also help improve lymphedema in patients unable to undergo manual lymph drainage.

The results of both conservative therapy and surgical therapy are dependent on the function of the remaining lymphatic vessels. In the present case, lymphoscintigraphy revealed good lymphatic vessel function;8 dilated collecting lymphatic vessels were evident in various locations during LVA,9,10 and a good postoperative outcome was achieved. An objective preoperative evaluation of lymphatic function is important, and

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lymphoscintigraphy, ICG lymphography,11 magnetic resonance lymphography,12 or another diagnostic imaging technique, must be performed. In the present case, rather than choosing between surgical therapy and conservative therapy, we treated the patient's lymphedema successfully using a

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combination of both. The normal tendency is to escalate to surgical therapy if conservative therapy fails. However, in patients, such as our patient, who have difficulty continuing conservative treatment for reasons such as cellulitis, intensifying conservative therapy after performing surgery may also be effective in controlling the disease. Combined conservative therapy and surgical therapy in lymphedema

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treatment may help reduce the frequency of cellulitis, reduce the circumference of the affected legs, and enable the use of less intensive conservative therapy. In addition to the use of conservative therapy, antibiotics, and surgical therapy

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independently decrease the frequency of cellulitis. Further studies are required to investigate the therapeutic efficacy of these treatments when used in combination. 4. Conclusion;

The combination of surgical treatment and conservative treatment is important for severe lymphedema treatment. Although conservative treatment is usually said to be

[Acknowledgement]

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the first-line, LVA can antecede in cases refractory to conservative treatment.

We are grateful for the total cooperation received from Eri Saeki, Izumi Masuda Sachiko Kimura,

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and Shigeru Harasawa (Saiseikai Kawaguchi general hospital).

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[Figure Legends]

Figure 1. Results of initial examination. The patient was not adherent to conservative therapy because of recurrent cellulitis. She presented with severe bilateral lower limb edema, generalized mild rubor, and lymphorrhea (leakage of lymphatic fluid from the dorsal surface of the thighs; 1–2 L/day).

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Figure2.

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Tc. Dilated collecting

Lymphoscintigraphy image 15 min after injection of

lymphatic vessels are present in both legs as far as the inguinal region, and the area

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within the pelvis is occluded.

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Figure 3. Indocyanine green (ICG) lymphography findings 8 h after injection of ICG.

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Dermal backflow is evident from the umbilical region to the distal end of the feet.

Figure 4. Immediate postoperative photographs. (a) First LVA. (b) Second LVA. The arrows show the sites of LVAs. N; Normal type of collecting lymphatic vessels, E; Ectasis

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type of collecting lymphatic vessels

Figure 5. Photograph of LVA. The collecting lymphatic vessel is dilated (ectatic;

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diameter, 0.8 mm). Ly; collecting lymph duct, V; Subcutaneous vein.

Figure 6. Results of clinical examination 6 months after the second LVA. Edema greatly improved, and cellulitis and lymphorrhea resolved.

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indocyanine green lymphographic findings with the conditions of collecting lymphatic

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[Figure Legends]

Figure 1. Results of initial examination. The patient was not adherent to conservative

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therapy because of recurrent cellulitis. She presented with severe bilateral lower limb edema, generalized mild rubor, and lymphorrhea (leakage of lymphatic fluid from the

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dorsal surface of the thighs; 1–2 L/day).

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Figure2.

99m

Tc. Dilated collecting

Lymphoscintigraphy image 15 min after injection of

lymphatic vessels are present in both legs as far as the inguinal region, and the area

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within the pelvis is occluded.

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Figure 3. Indocyanine green (ICG) lymphography findings 8 h after injection of ICG.

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Dermal backflow is evident from the umbilical region to the distal end of the feet.

Figure 4. Immediate postoperative photographs. (a) First LVA. (b) Second LVA. The arrows show the sites of LVAs. N; Normal type of collecting lymphatic vessels, E; Ectasis

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type of collecting lymphatic vessels

Figure 5. Photograph of LVA. The collecting lymphatic vessel is dilated (ectatic;

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diameter, 0.8 mm). Ly; collecting lymph duct, V; Subcutaneous vein.

Figure 6. Results of clinical examination 6 months after the second LVA. Edema greatly improved, and cellulitis and lymphorrhea resolved.

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