The successful use of lipectomy in the management of airway obstruction in a woman with HIV-associated lipodystrophy

Abstract

Lipodystrophy is a common complication of highly active antiretroviral therapy and is associated with significant comorbidities. Altered fat distribution, particularly lipohypertrophy of the dorsal cervical fat pad is associated with reduced quality of life as well as medical complications. We report the rare case of a patient with airway obstruction secondary to HIV-associated lipodystrophy. Ultrasound-assisted liposuction was successfully performed to relieve her airway obstruction and to facilitate a tracheostomy. To the best of our knowledge, this is the first documented case of its kind. We also provide a brief review of the literature on the current management options for HIV-associated lipodystrophy.

Background

Highly active antiretroviral therapy (HAART) has dramatically decreased mortality associated with HIV.¹ Unfortunately, HAART is associated with multiple metabolic disorders and causes prominent lipodystrophy in 2–13% of patients.² Lipodystrophy includes both lipohypertrophy and lipoatrophy. This can result in disfiguring fat distribution favouring the neck, trunk and breasts; and can result in significant complications. In the majority of cases the aetiology is multifactorial but often attributed to HAART, particularly with the use of protease inhibitors, nucleotide reverse-transcriptase inhibitors, and non-nucleoside reverse transcriptase inhibitors.^{3 4} Evidence suggests this may be related to the structural analogy at the molecular level between HAART medications and proteins involved in lipid metabolism.⁵ Furthermore, this could potentially explain some of the comorbidities often associated with HAART.

In this report, we describe the use of a common elective surgical procedure for the management of a rare case of airway obstruction leading to respiratory failure in the setting of HIV-associated lipodystrophy. We discuss our patient's surgical management, and provide a brief literature review on current medical and surgical treatment options for HIV-associated lipodystrophy.

Case presentation

A 57-year-old woman was referred to plastic surgery regarding further management of her airway obstruction. The patient's medical history included chronic obstructive pulmonary disease (COPD), obstructive sleep apnoea, diabetes mellitus, hyperlipidaemia, and HIV diagnosed over 15 years ago. Her HAART regimen included darunavir, ritonavir, emtricitabine and tenofovir.

She initially presented to the emergency department in acute respiratory distress. She was found to be afebrile and tachypnoeic, with oxygen saturation of 88% on nasal cannula. A chest X-ray at that time revealed low lung volumes. A trial of non-invasive positive-pressure ventilation did not improve her condition. She was intubated and admitted to the intensive care unit for respiratory failure. The clinical picture and radiological investigation was consistent with an acute exacerbation of COPD. This was compounded by her significant lipohypertrophy which we believe placed an additional obstructive load on her airway. Over the next several weeks, multiple attempts to extubate the patient were unsuccessful. Tracheostomy was recommended but not performed initially due to the high risk of mortality in this patient with profound cervical lipohypertrophy.

Owing to the complexity of her case, plastic surgery was consulted. On examination, she had massive lipohypertrophy of the anterior, lateral and dorsal cervical fat pads (figure 1).

Figure 1.

Preoperative appearance of the patient prior to surgery. (A) Lipohypertrophy of the anterior and lateral cervical fat pads. (B) Lipohypertrophy of the dorsal cervical fat pad (‘buffalo hump’).

Treatment

Both ultrasonic liposuction and open neck lipectomy were considered. The patient required posterior as well as anterior correction because the large posterior collection of adipose tissue was driving the chin/neck into the flexed position, further compromising the airway. Open technique was fraught with large blood loss, technical closure issues and prolonged suction catheter drainage of the wound. Ultrasound-assisted liposuction was deemed to be safer. Any temporary compression associated with fluid insufflations used to accomplish ultrasonic liposuction would be compensated for with aggressive liposuction removing fat and fluid. Postoperative oedema and induration would be controlled with intravenous steroids and continued temporary intubation. Given these facts, the decision was made to perform ultrasound-assisted liposuction to help relieve her airway obstruction and to facilitate a tracheostomy. Initially, she was placed in the prone position in an attempt to reduce the dorsal cervical fat pads. However, her oxygen saturation deteriorated which provided further evidence of her tenuous respiratory condition. Placement in the left lateral decubitus position returned her oxygen saturation to 95%. Local anaesthetic was applied prior to cannula insertion. Tumescent infiltration with epinephrine 1:100 000 in Ringers Lactate solution was used. 3100 cc of infiltrate was infused into the posterior neck, and 4000 cc of aspirate was suctioned. She was placed into the supine position and 1550 cc of infiltrate was infused into the anterior neck, and 1500 cc of aspirate was suctioned (figure 2). Estimated blood loss was minimal. The total procedure time was 6 h.

Figure 2.

Anterior, dorsal cervical and back fat pads after ultrasound-assisted lipectomy. (A) Anterior and lateral fat pads. (B) Dorsal cervical and back fat pads.

Outcome and follow-up

The patient remained intubated immediately postoperatively. Her postoperative course was complicated by a cerebral vascular accident. On postoperative day 12, an elongated XLT tracheostomy tube was successfully placed for long-term respiratory support. She was discharged 1 week later to a skilled nursing facility and has performed well. At 6 months postoperative, the patient is near baseline, alert and breathing without the need for ventilator support (figure 3).

Figure 3.

The patient at 6 months status post ultrasound-assisted lipectomy demonstrating improved contour of the neck. (A) Anterior and (B) lateral.

Discussion

In our broad review of the literature, use of liposuction for non-elective procedures is uncommon but has been described for cases such as haematoma evacuation.⁶ In a similar case to our patient's, cervical fat excision was performed to facilitate tracheostomy placement in an HIV-negative, morbidly obese man.⁷ To the best of our knowledge, there are two documented reports of lipodystrophy resulting in airway obstruction. Press et al⁸ described a HIV-positive man with comorbid COPD who unfortunately succumbed to pneumonia several weeks after his initial presentation. Neither lipectomy nor tracheostomy was part of his management. The second case, involved a HIV-negative woman with MERRF syndrome (myoclonic epilepsy with ragged red-fibers), an extremely rare genetic disorder of mitochondrial oxidative phosphorylation that manifested with idiosyncratic lipodystrophy causing life-threatening respiratory obstruction. She was successfully managed with multistaged surgical excision and suction-assisted lipoplasty.⁹

Current treatments for HIV-associated lipodystrophy include medical and surgical options. When possible, switching the use of a current agent, particularly stavudine or zidovudine, to an alternative that has a lesser degree of association with lipodystrophy can be attempted.^{10 11} There are limited medical options specifically available for HIV-associated lipodystrophy, with many focused instead on the management of comorbid metabolic disturbances such as diabetes and hyperlipidaemia.¹² However, there is evidence that suggests metformin may be beneficial at reducing visceral and subcutaneous abdominal fat.^{13 14} Also, tesamorelin, a recombinant growth hormone-releasing factor analogue, demonstrates moderate effectiveness in decreasing visceral adipose tissue.¹⁵ Lastly, there may be some benefit with other agents such as anabolic steroids, didehydroepiandrosterone, naltrexone and indomethacin.¹⁶

Surgical options are dependent on the distribution and the presence of lipoatrophy versus lipohypertrophy. The most common site of lipoatrophy is the face. Surgical options include autologous fat transfer, dermis-fat grafts, flaps, rhytidectomy, and soft tissue fillers such as calcium hydroxylapatite.^{17 18} Several studies have demonstrated that elective surgical management of HIV-associated lipohypertrophy with ultrasound-assisted liposuction or conventional suction-assisted lipectomy were successful in reducing the dorsal cervical fat pad with minimal morbidity.^19–21 While the literature on long-term efficacy is limited, recurrence has been noted in several patients.^{22 23} Further research regarding this subject area is needed.

In addition to the obvious physical and medical benefits of treatment, previous studies have demonstrated the impact of lipodystrophy on medication adherence, patient self-esteem, and quality of life.^{24 25} This case demonstrates that ultrasound-assisted liposuction can be considered as part of the management for airway obstruction secondary to HIV-associated lipodystrophy. For HIV-positive patients with significant lipodystrophy and comorbid conditions such as COPD, assessment of pulmonary function should be carefully monitored and elective correction of lipodystrophy should be considered to prevent the potential for respiratory failure and help improve quality of life.

Learning points.

• Lipodystrophy is a known complication of highly active antiretroviral therapy and can rarely present with airway obstruction, particularly in the setting of comorbid lung disease.

• For patients who do not manifest obstructive symptoms while awake, polysomnography should be considered to diagnose or rule out HIV-associated lipodystrophy as a cause of sleep apnoea.

• Assessment of pulmonary function is suggested in patients with significant lipodystrophy, and elective correction should be considered to prevent respiratory complications.

• Surgical lipectomy can be considered as part of the management of airway obstruction in these patients, and may help to facilitate an otherwise difficult tracheostomy.