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Massive localized lymphedema of the mons: the pi-cut technique

DOI:10.1007/s00238-016-1270-y
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Hubert B. Shih
Hubert B. Shih
Hubert B. Shih
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Rahim Nazerali
Rahim Nazerali
Rahim Nazerali
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Anandev Gurjala
Anandev Gurjala
Anandev Gurjala
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Leila Jazayeri
Leila Jazayeri
Leila Jazayeri
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Abstract and Figures

Massive localized lymphedema (MLL) is a form of secondary lymphedema caused by obstruction of lymphatic flow seen in morbidly obese patients. Common areas for the large mass include the abdomen, suprapubic region, scrotum, and thigh. Skin changes and the sheer size cause significant morbidity. Described as a benign lymphoproliferative overgrowth, MLL if left untreated can progress to angiosarcoma. The key to resection is minimizing blood loss and the key to reconstruction is to reconstruct utilizing the principles of vulvar esthetic subunits. For hemostasis, a tourniquet is used around the base of the mass and infiltration with wetting solution is injected. Reconstruction with the “pi-cut” technique, so named because of its resemblance to the mathematical symbol of pi, allows closure along the vulvar esthetic subunits. To illustrate our surgical technique, we present a case of MLL of the mons veneris in a 53-year-old woman with a BMI (body mass index) of 78 kg/m2. The specimen weighed approximately 17.5 kg, the heaviest MLL of the mons veneris described in the literature. Our resection technique and pi-cut closure technique allowed for minimal blood loss and postoperative morbidity and an esthetic closure respecting the vulvar esthetic subunits. MLL of the mons veneris can be successfully and safely resected with an esthetic result using our technique. Level of Evidence: Level V, therapeutic study.
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IDEAS AND INNOVATIONS
Massive localized lymphedema of the mons: the pi-cut technique
Hubert B. Shih
1
&Rahim Nazerali
1
&Anandev Gurjala
1
&Leila Jazayeri
1
&
Gordon K. Lee
1
Received: 31 July 2016 /Accepted: 26 December 2016 /Published online: 16 January 2017
#Springer-Verlag Berlin Heidelberg 2017
Abstract Massive localized lymphedema (MLL) is a form of
secondary lymphedema caused by obstruction of lymphatic
flow seen in morbidly obese patients. Common areas for the
large mass include the abdomen, suprapubic region, scrotum,
and thigh. Skin changes and the sheer size cause significant
morbidity. Described as a benign lymphoproliferative over-
growth, MLL if left untreated can progress to angiosarcoma.
The key to resection is minimizing blood loss and the key to
reconstruction is to reconstruct utilizing the principles of vulvar
esthetic subunits. For hemostasis, a tourniquet is used around
the base of the mass and infiltration with wetting solution is
injected. Reconstruction with the Bpi-cut^technique, so named
because of its resemblance to the mathematical symbol of pi,
allows closure along the vulvar esthetic subunits. To illustrate
our surgical technique, we present a case of MLL of the mons
veneris in a 53-year-old woman with a BMI (body mass index)
of 78 kg/m2. The specimen weighed approximately 17.5 kg,
the heaviest MLL of the mons veneris described in the litera-
ture. Our resection technique and pi-cut closure technique
allowed for minimal blood loss and postoperative morbidity
and an esthetic closure respecting the vulvar esthetic subunits.
MLL of the mons veneris can be successfully and safely
resected with an esthetic result using our technique.
Level of Evidence: Level V, therapeutic study.
Keywords Lymphedema .Obesity .Morbid .Reconstructive
surgical procedures
Introduction
Massive localized lymphedema (MLL) is a benign over-
growth of lymphoproliferative tissue and a known complica-
tion of morbid obesity. The etiology has not been totally de-
fined, but it is thought to be due to mechanical alterations in
lymph drainage due to obesity leading to lymphedema [1].
First described by Farshid and Weiss in 1998, it was termed
massive localized lymphedema due to the clinical presentation
and morphology. The lesion was noted to be histologically
similar to well-differentiated liposarcoma; hence, the term
pseudosarcoma is occasionally used [2].
Patients with MLL usually present with a large painless
mass that has been slowly growing for several years.
Ultimately, the size causes significant functional limitations.
The thickened, woody, and excoriated skin can also cause
significant morbidity including chronic wounds and infec-
tions. In 2013, Chopra et al. surveyed all the MLL cases pre-
sented in the literature and found the average BMI to be 61 kg/
m2 with the most common location being the thigh. Other
common areas include the lower abdomen, the mons, and
the scrotum. The mons is unique in that reconstruction after
resection needs to take into account the vulvar esthetics. The
technique proposed here offers resection with key attention to
minimizing blood loss, and the reconstruction method re-
spects the vulvar subunits. A case utilizing these principles
is presented. Overall, five cases of MLL of the mons are re-
ported in the literature; the case presented here is the heaviest
specimen from a female on record [3].
Surgical technique
Because patients with MLL suffer from a severe reduction in
quality of life and problems with hygiene, as well as concerns
*Hubert B. Shih
hshih@stanford.edu
1
Department of Surgery, Division of Plastic and Reconstructive
Surgery, Stanford Hospital and Clinics, 770 Welch Road, Suite 400,
Palo Alto, CA 94304, USA
Eur J Plast Surg (2017) 40:343–346
DOI 10.1007/s00238-016-1270-y
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Massive Localized Lymphedema in the Morbidly Obese
Article
We report 14 cases of a soft tissue lesion in the limbs of morbidly obese adults that presents as a large mass and histologically simulates well-differentiated liposarcoma (WDL). Based on its distinctive clinical setting and morphologic identity to diffuse lymphedema we have termed this process massive localized lymphedema (MLL). All cases occurred in morbidly obese adults (mean weight 372 lbs; mean age 47 years). Women predominated (9 women; 5 men). The lesions affected the proximal medial aspect of the extremities (12 thigh; 2 arm) and were unilateral in all but two patients. Etiologically significant antecedent events include ipsilateral axillary lymphadenectomy in both patients with arm lesions, chronic lymphedema resulting from vein-stripping 10 years prior in one patient, inguinal lymphadenectomy for anal carcinoma in another patient, and significant blunt trauma to the inner thigh during a motor vehicle accident in a third patient. The tumors were long standing (1-10 years) and extremely large (mean size 33.4 cm, 7408 g). Clinically, they were diffuse, ill-defined masses that histologically consisted of lobules of mature fat interrupted by expanded connective tissue septa. The constituents of the septa were fine, fibrillary collagen, edema fluid, and uniformly distributed fibroblasts. Clusters of capillaries were frequently found at the interface between fat and connective tissue. The widened septa simulated the fibrous bands of sclerosing WDL, but MLL lacks the degree of nuclear atypia seen in the former. The consistent clustering of reactive vessels at the interface between the fat and fibrous tissue also contrasted with WDL. Six patients experienced persistent or recurrent lesions within 10 months to 10 years. No aggressive growth or histologic progression was observed during this time, however. Awareness of the features of MLL is important to avoid misclassification of this reactive lesion with WDL.
Going in the Wrong Direction With Monsplasty
Article
Two recent articles in Aesthetic Surgery Journal —by Alter1 and Bloom et al2—have addressed techniques for monsplasty, combined with either labia majora reduction or abdominoplasty, for massive weight loss patients. The techniques by Alter and Bloom et al also have a common theme: defatting and vertical pull.1,2 The problem with these techniques is that the defect is not vertical excess. If anything, the mons that needs monsplasty is short and fat and has a width greater than its height. When a vertical lift technique is used, the hair-bearing skin is pulled up, elongating the mons and giving the appearance that the tissue has been pulled too high. Using the subunit principle, there are defining landmarks to the mons—it is a triangle with boundaries. These boundaries are the lateral junction with the thigh, the superior boundary with the abdomen …
Massive Localized Lymphedema Revisited: A Quickly Rising Complication of the Obesity Epidemic
Article
Massive localized lymphedema (MLL) is a rising and potentially fatal complication of the obesity epidemic. Described as a benign lymphoproliferative overgrowth of obese patients, MLL is a form of secondary lymphedema, caused by the obstruction of lymphatic flow, with characteristic clinical and histological presentation. Patients have a large mass with classic skin changes often accompanied by lymphatic weeping that require complex reconstruction. Although oftentimes benign, if left untreated, MLL can progress to angiosarcoma, further supporting the need for more research into MLL and its sequelae. We present a unique case of MLL of the mons pubis in a 52-year-old man with a body mass index of 75.7 kg/m. The literature was comprehensively reviewed with a total of 65 cases of MLL being described, 9 of which resulted in angiosarcoma (10.3% of all cases), 6 of which resulted in death (9.2% of all cases). We found a female predominance of 1.24 to 1, an average weight of 183 kg, and a 48.5% majority of cases in the thigh.
Massive Localized Lymphedema Review of an Emerging Problem and Report of a Complex Case in the Mons Pubis
Article
Massive localized lymphedema (MLL) is an emerging complication of the obesity epidemic. Caused by the obstruction of lymphatics, MLL presents as a giant swelling, with characteristic skin changes, and often lymphatic weeping. MLL has also been called "pseudosarcoma" because of its morphologic and pathologic similarity to sarcoma. Left untreated, MLL can degenerate into angiosarcoma. We present a case of MLL of the mons pubis in a 40-year-old man with a body mass index of 69. The literature is reviewed and an additional 40 cases of MLL are described. We found a female predominance of 1.6 to 1, an average weight of 421 lbs, and a 58% majority of cases in the thigh.
Massive Localized Lymphoedema
Article
A case report of massive localized lymphoedema in a morbidly obese 50-year-old woman and review of the literature.
Abdominal Contour Surgery: Treating All Aesthetic Units, Including the Mons Pubis
Article
Background: Many patients who seek “abdominal” contour surgery also desire improvement of areas of the torso adjacent to the abdomen, such as the flanks and back rolls. Objective: We propose a classification of the abdomen into distinct aesthetic units and report on a series of cases in which this classification was used, with special emphasis on one such unit, the mons pubis. Methods: A series of 400 consecutive patients were evaluated according the proposed designation of aesthetic units. Lipoplasty was performed as described in previous studies. A subset of 6 patients who specifically desired improvement of the mons pubis were treated by lipoplasty and/or horizontal or vertical wedge incision. Results: Most patients who received treatment according to aesthetic unit were satisfied with their outcomes. Two of the patients who sought improvement of the mons pubis specifically requested additional procedures. Conclusions: An approach to abdominal contour surgery that includes consideration of all related aesthetic units can achieve more proportional, harmonious results and increase patient satisfaction.
Massive localized lymphedema in the morbidly obese: a histologically distinct reactive lesion simulating liposarcoma
Article
We report 14 cases of a soft tissue lesion in the limbs of morbidly obese adults that presents as a large mass and histologically simulates well-differentiated liposarcoma (WDL). Based on its distinctive clinical setting and morphologic identity to diffuse lymphedema we have termed this process massive localized lymphedema (MLL). All cases occurred in morbidly obese adults (mean weight 372 lbs; mean age 47 years). Women predominated (9 women; 5 men). The lesions affected the proximal medial aspect of the extremities (12 thigh; 2 arm) and were unilateral in all but two patients. Etiologically significant antecedent events include ipsilateral axillary lymphadenectomy in both patients with arm lesions, chronic lymphedema resulting from vein-stripping 10 years prior in one patient. inguinal lymphadenectomy for anal carcinoma in another patient, and significant blunt trauma to the inner thigh during a motor vehicle accident in a third patient. The tumors were long standing ( I-IO years) and extremely large (mean size 33.4 cm, 7408 g). Clinically, they were diffuse, ill-defined masses that histologically consisted of lobules of mature fat interrupted by expanded connective tissue septa. The constituents of the septa were fine, fibrillary collagen, edema fluid, and uniformly distributed fibroblasts. Clusters of capillaries were frequently found at the interface between fat and connective tissue. The widened septa simulated the fibrous bands of sclerosing WDL, but MLL lacks the degree of nuclear atypia seen in the former. The consistent clustering of reactive vessels at the interface between the fat and fibrous tissue also contrasted with WDL. Six patients experienced persistent or recurrent lesions within 10 months to 10 years. No aggressive growth or histologic progression was observed during this time, however. Awareness of the features of MLL is important to avoid misclassification of this reactive lesion with WDL.