Morbihan disease: A diagnostic dilemma: two cases with successful resolution

Donthi, Deepak; Nenow, Joseph; Samia, Arthur M; Phillips, Charles M.; Papalas, John A.; Prenshaw, Karyn

doi:10.1177/2050313x211023655

Cited by 13 publications

(19 citation statements)

References 15 publications

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“…Morbihan syndrome is so uncommon that it is only reported in case studies [6,[11][12][13]. No previous case reports link the syndrome to TN or show improvement in symptoms following treatment of TN with SRS.…”

Section: Discussionmentioning

confidence: 99%

Treatment of Trigeminal Neuralgia With Stereotactic Radiosurgery Improved Symptoms of Morbihan Syndrome

Burgess¹,

Mula‐Hussain²,

Malone³

2021

Cureus

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Trigeminal neuralgia (TN) is a neuropathic pain disorder characterized by paroxysmal pain in the maxillary and mandibular regions of the face. Morbihan syndrome is a disease that classically presents with dermatologic findings, including progressive facial edema and erythema. There are no previous reports of the onset of trigeminal neuralgia with Morbihan syndrome or previous reports describing improvement in symptoms of Morbihan syndrome with treatment of trigeminal neuralgia. We describe the case of a 62-yearold female who presented with trigeminal neuralgia and shortly thereafter developed significant facial edema and was diagnosed with Morbihan syndrome. The neuralgia was refractory to medical management and was effectively treated with stereotactic radiosurgery (SRS). This coincided with an improvement in her Morbihan syndrome that is now controlled following stereotactic radiosurgery and continued lymphatic massage.

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Section: Discussionmentioning

confidence: 99%

Treatment of Trigeminal Neuralgia With Stereotactic Radiosurgery Improved Symptoms of Morbihan Syndrome

Burgess¹,

Mula‐Hussain²,

Malone³

2021

Cureus

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“…MD, also called rosaceous lymphoedema, is clinically characterized by firm, non-pitting edema and erythema. Symptoms appear throughout the middle and upper thirds of the face [ 4 , 8 , 11 ]. The pathogenesis of MD is unclear.…”

Section: Discussionmentioning

confidence: 99%

Lymphaticovenular anastomosis for Morbihan disease: a case report

Hong

Lee

et al. 2023

Arch Craniofac Surg

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Morbihan disease (MD) is a very rare condition characterized by rosaceous or erythematous lymphedema on the upper twothirds of the face. A definitive management strategy for MD is lacking, and treatment is challenging. Herein, we present a case of persistent bilateral eyelid edema treated by lymphaticovenular anastomosis (LVA) and lymph node-vein bypass surgery. The patient experienced persistent bilateral eyelid edema. Indocyanine green lymphography was performed, and the diagnosis of bilateral facial lymphedema was confirmed. On the right side, a preauricular lymphatic vessel was anastomosed to a vein. On the left side, lymphostomy on the preauricular lymph node was done, with anastomosis to the transected proximal end of the concomitant vein of the transverse facial artery. Furthermore, a preauricular lymphatic vessel was anastomosed to a vein. Eyelid edema decreased and progressively improved on both sides. The outcome of this case suggests that LVA and lymph node-vein bypass surgery are appropriate for treating persistent eyelid edema related to MD.

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“…This case report emphasizes 2 important points about Morbihan disease: it is difficult to diagnose and it is often refractory to typical therapies. 1 , 4 With regard to diagnosis, a thorough workup, including biopsy and clinicopathologic correlation, is recommended to assist in differentiating Morbihan disease from other systemic causes. As for treatment, this case suggests that oculoplastic surgery, such as blepharoplasty, offers a potential short-term therapeutic alternative for patients with recalcitrant Morbihan disease involving the eyelids.…”

Section: Discussionmentioning

confidence: 99%