Ovid Therapeutics Reports Second Quarter 2018 Financial Results and Highlights Recent Clinical Progress -- OV101 in Angelman syndrome: plan to initiate an open-label extension study (ELARA) in the fourth quarter of 2018

Ovid Therapeutics Reports Second Quarter 2018 Financial Results and Highlights Recent Clinical Progress

-- OV101 in Angelman syndrome: plan to initiate an open-label extension study (ELARA) in the fourth quarter of 2018 and discuss with regulatory authorities next steps for a registrational pathway based on positive Phase 2 STARS data --

-- OV101 in Fragile X syndrome: topline data from Phase 2 ROCKET trial in males with Fragile X syndrome expected in 2019 --

-- OV935/TAK-935 in rare developmental and epileptic encephalopathies: data from Phase 1b/2a study expected in fourth quarter of 2018; ENDYMION, open-label extension study, now enrolling patients --

NEW YORK - August 9, 2018 - Ovid Therapeutics Inc. (NASDAQ: OVID), a biopharmaceutical company committed to developing medicines that transform the lives of people with rare neurological disorders, today reported financial results for the second quarter ended June 30, 2018 and provided an overview of the company's recent clinical progress.

"Earlier this week we announced positive topline data from the Phase 2 STARS trial in Angelman syndrome, the first industry-sponsored, randomized, double-blind, placebo-controlled study with a hierarchy of prespecified endpoints," said Jeremy Levin, DPhil, MB, BChir, chairman and chief executive officer of Ovid Therapeutics. "OV101 is the first investigational medicine to demonstrate a significant clinical effect in overall symptomatology (using the CGI-I clinician- reported measurement) since the condition was identified more than 50 years ago. Importantly, CGI-I was the first measure on the prespecified endpoint analysis in the STARS trial. This resulted in a robust design and helped ensure that investigators captured the impact of OV101 on the totality of global neurological deficits and symptomatology. Angelman syndrome is a complex disorder that impacts all areas of the brain and we believe CGI-I is an appropriate assessment tool for overall Angelman syndrome symptomatology. Having also identified what we believe is the optimal dose of once-daily OV101 to bring forward, preparations are underway to discuss with regulatory authorities the next steps for a registrational pathway."

Dr. Levin continued, "Beyond the STARS trial, the Phase 2 ROCKET trial with OV101 in adolescents and young adult males with Fragile X syndrome is initiated, and we plan to start the accompanying SKY ROCKET non-drug study in the third quarter of 2018. Our collaboration with Takeda for the development of OV935/TAK-935 in developmental and epileptic

encephalopathies (DEE) also continues to advance in the clinic. We look forward to a busy rest of the year, as we anticipate topline data from our Phase 1b/2a clinical trial of OV935 in adults with DEE, as well as the initiation of ELEKTRA and ARCADE, two Phase 2 studies in pediatric DEE patients."

Recent Progress and Upcoming Milestones

OV101 for Angelman Syndrome

OV101 for Fragile X Syndrome

OV935 for Rare Developmental and Epileptic Encephalopathies

OV329 for Treatment-Resistant Epilepsy

Second Quarter 2018 Financial Results

Research and development expenses were $8.1 million for the second quarter of 2018, as compared to $6.1 million for the same period in 2017. The increase of $2.0 million was primarily due to an increase in preclinical and development expenses.

General and administrative expenses were $5.1 million for the second quarter of 2018, as compared to $4.2 million for the same period in 2017. The increase was primarily due to higher payroll and payroll-related expenses due to growth in headcount as the company expanded its operations, and an increase in professional fees associated with operating as a public company.

Net loss was $12.9 million, or net loss per share of $0.53, for the second quarter of 2018, as compared to a net loss of $10.2 million, or net loss per share of $0.57, for the same period in 2017.

As of June 30, 2018, cash, cash equivalents and short-term investments totaled $62.6 million.

OV101 (gaboxadol) is believed to be the only delta ( )-selective GABAA receptor agonist in development and the first investigational medicine to specifically target the disruption of tonic inhibition, a central physiological process of the brain that is thought to be the underlying cause of certain neurodevelopmental disorders. OV101 has demonstrated in laboratory studies and animal models to selectively activate the -subunit of GABAA receptors, which are found in the extrasynaptic space (outside of the synapse), and thereby impact neuronal activity through tonic inhibition.

Ovid is developing OV101 for the treatment of Angelman syndrome and Fragile X syndrome to potentially restore tonic inhibition and relieve several of the symptoms of these disorders. In preclinical studies, it was observed that OV101 improved symptoms of Angelman syndrome and Fragile X syndrome. In the STARS Phase 2 trial, OV101 showed a favorable safety profile, was well tolerated, and showed a significant improvement on the Clinical Global Impressions-Improvement (CGI-I) scale in individuals with Angelman syndrome. Gaboxadol has previously been tested in over 4,000 patients (1,000+ patient-years of exposure) and was observed to have favorable safety and bioavailability profiles.

The U.S. Food and Drug Administration (FDA) has granted orphan drug and Fast Track designations for OV101 for both the treatment of Angelman syndrome and Fragile X syndrome. The U.S. Patent and Trademark Office has granted Ovid patents directed to methods of treating Angelman syndrome and Fragile X syndrome using OV101. The issued patents expire in 2035 without regulatory extensions.

OV935/TAK-935 is a potent, highly-selective, first-in-class inhibitor of the enzyme cholesterol 24-hydroxylase (CH24H) being investigated as an anti-epileptic drug (AED). CH24H is predominantly expressed in the brain, where it plays a central role in cholesterol homeostasis. CH24H converts cholesterol to 24-hydroxycholesterol (24HC), which then exits the brain into the blood plasma circulation. Glutamate is one of the main neurotransmitters in the brain and has been shown to play a role in the initiation and spread of seizure activity. Recent literature indicates CH24H is involved in over-activation of the glutamatergic pathway through modulation of the NMDA channel, implying its potential role in central nervous system diseases such as epilepsy. Ovid and Takeda believe that OV935's novel mechanism of action may potentially treat rare epilepsies by inhibiting CH24H to decrease 24HC levels, effectively decreasing glutamate hyperactivity. This mechanism of action may be especially important in CDD and Dup15q since the NMDA receptor-mediated synaptic transmission underlies the pathological mechanisms of these syndromes. To Ovid and Takeda's knowledge, OV935 is the only molecule with this mechanism of action in clinical development. OV935 is an investigational drug, not approved for commercial use.

OV935 has successfully completed four Phase 1 clinical studies, which have assessed tolerability, PK and target engagement at doses believed to be therapeutically relevant. In preclinical models, a novel proprietary PET ligand was used to determine target occupancy of OV935 in the brain. OV935 is being co-developed by Ovid and Takeda Pharmaceutical Company Limited.

OV329 is a preclinical compound being developed by Ovid Therapeutics for epilepsy and other neurologic disorders, as part of the company's epilepsy portfolio. OV329 functions by inactivating GABA aminotransferase (GABA-AT), a key metabolic enzyme of the brain's major inhibitory neurotransmitter, GABA. By inhibiting the metabolism of GABA, OV329 leads to increased intracellular concentrations of GABA. Given that epilepsy is characterized by excessive neuronal excitation, the enhanced release of GABA may suppress this excitatory signaling and may reduce seizures. GABA-AT is a well-validated target for treatment-resistant epilepsy and has applications in multiple seizure types such as refractory complex partial seizures and infantile spasms.

About Ovid Therapeutics

Ovid Therapeutics (NASDAQ: OVID) is a New York-based biopharmaceutical company using its BoldMedicine approach to develop medicines that transform the lives of people with rare neurological disorders. Ovid has a broad pipeline of first-in-class medicines. The company's lead investigational medicine, OV101, is in development for the treatment of Angelman syndrome and Fragile X syndrome. Ovid is also developing OV935 in collaboration with Takeda Pharmaceutical Company Limited for the treatment of rare developmental and epileptic encephalopathies (DEE).

For more information on Ovid, please visit http://www.ovidrx.com/.

Forward-Looking Statements

This press release includes certain disclosures that contain "forward-looking statements," including, without limitation, statements regarding (i) the initiation, progress, timing, scope and results of clinical trials for Ovid's product candidates, (ii) the company's preclinical and clinical development plans, (iii) the development of new therapies for previously unidentified disorders, (iv) the number of patients to be enrolled, (v) the timing of reporting of clinical data regarding Ovid's product candidates, (vi) the presentation of scientific date at scientific meetings; and (vii) the approval or registration of Ovid's product candidates by regulatory authorities. You can identify forward-looking statements because they contain words such as "will," "believes" and "expects." Forward-looking statements are based on Ovid's current expectations and assumptions. Because forward-looking statements relate to the future, they are subject to inherent uncertainties, risks and changes in circumstances that may differ materially from those contemplated by the forward-looking statements, which are neither statements of historical fact nor guarantees or assurances of future performance. Important factors that could cause actual results to differ materially from those in the forward-looking statements are set forth in Ovid's filings with the Securities and Exchange Commission, under the caption "Risk Factors." Ovid assumes no obligation to update any forward-looking statements contained herein to reflect any change in expectations, even as new information becomes available.

Condensed Statements of Operations

1Working capital defined as current assets less current liabilities

Ovid Therapeutics Inc.

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